Obesity not only increases a patient’s lifetime risk of numerous chronic conditions, such as diabetes, heart disease and kidney disease, but it also is a major health issue during pregnancy. Women who are obese in pregnancy have a significantly higher chance of developing adverse perinatal outcomes and experiencing various complications that affect both their health and that of their babies.

With an ever-increasing population of overweight and obese women of reproductive age, as key caregivers for women, we must reexamine our approaches and do much more than microfocusing on a woman’s pre- and postdelivery health. We must play a more active role in helping our patients establish and maintain a healthy lifestyle – one that will help ward off and reduce the incidence of this concerning condition.

Over the previous two Master Class installments on obstetrics, we discussed the extent of the obesity epidemic and its link to diabetes, the alarming number of infants, children, and adolescents who are obese, and the implications of these societal and medical trends for ob.gyns.

In the July Master Class, we discussed the importance of appropriately counseling patients on healthy weight gain and physical activity in pregnancy. Because ob.gyns. may be the only health care professionals that many women may see, it is becoming more important that we help our patients and their children attain and maintain positive health and well-being.

In our September installment, Dr. Thomas R. Moore looked at obesity trends through the lens of the Barker Hypothesis, which got us thinking more than 3 decades ago about the role of intrauterine environment in short- and long-term health of offspring. Dr. Moore discussed how obesity in pregnancy appears to program offspring for downstream cardiovascular risk in adulthood.

He told us that we must not only liberally treat gestational diabetes and optimize glucose control during pregnancy, but, most importantly, we also must emphasize to women the importance of having healthy weights at the time of conception.

This month’s Master Class examines this latter concept in more depth. Dr. Patrick Catalano, professor in the department of obstetrics and gynecology and director of the Center for Reproductive Health at MetroHealth Medical Center, Case Western Reserve University, Cleveland, has been at the forefront of research on the physiologic impact of obesity on the placenta and the fetus, and on approaches for addressing maternal obesity and improving perinatal outcomes.

Dr. Catalano explains here why weight loss before pregnancy appears to be important for preventing adverse perinatal outcomes and breaking the intergenerational transfer of obesity.

Dr. Reece, who specializes in maternal-fetal medicine, is vice president for medical affairs at the University of Maryland, Baltimore, as well as the John Z. and Akiko K. Bowers Distinguished Professor and dean of the school of medicine. Dr. Reece said he had no relevant financial disclosures. He is the medical editor of this column. Contact him at [email protected].

Obesity not only increases a patient’s lifetime risk of numerous chronic conditions, such as diabetes, heart disease and kidney disease, but it also is a major health issue during pregnancy. Women who are obese in pregnancy have a significantly higher chance of developing adverse perinatal outcomes and experiencing various complications that affect both their health and that of their babies.

With an ever-increasing population of overweight and obese women of reproductive age, as key caregivers for women, we must reexamine our approaches and do much more than microfocusing on a woman’s pre- and postdelivery health. We must play a more active role in helping our patients establish and maintain a healthy lifestyle – one that will help ward off and reduce the incidence of this concerning condition.

Over the previous two Master Class installments on obstetrics, we discussed the extent of the obesity epidemic and its link to diabetes, the alarming number of infants, children, and adolescents who are obese, and the implications of these societal and medical trends for ob.gyns.

In the July Master Class, we discussed the importance of appropriately counseling patients on healthy weight gain and physical activity in pregnancy. Because ob.gyns. may be the only health care professionals that many women may see, it is becoming more important that we help our patients and their children attain and maintain positive health and well-being.

In our September installment, Dr. Thomas R. Moore looked at obesity trends through the lens of the Barker Hypothesis, which got us thinking more than 3 decades ago about the role of intrauterine environment in short- and long-term health of offspring. Dr. Moore discussed how obesity in pregnancy appears to program offspring for downstream cardiovascular risk in adulthood.

He told us that we must not only liberally treat gestational diabetes and optimize glucose control during pregnancy, but, most importantly, we also must emphasize to women the importance of having healthy weights at the time of conception.

This month’s Master Class examines this latter concept in more depth. Dr. Patrick Catalano, professor in the department of obstetrics and gynecology and director of the Center for Reproductive Health at MetroHealth Medical Center, Case Western Reserve University, Cleveland, has been at the forefront of research on the physiologic impact of obesity on the placenta and the fetus, and on approaches for addressing maternal obesity and improving perinatal outcomes.

Dr. Catalano explains here why weight loss before pregnancy appears to be important for preventing adverse perinatal outcomes and breaking the intergenerational transfer of obesity.

Dr. Reece, who specializes in maternal-fetal medicine, is vice president for medical affairs at the University of Maryland, Baltimore, as well as the John Z. and Akiko K. Bowers Distinguished Professor and dean of the school of medicine. Dr. Reece said he had no relevant financial disclosures. He is the medical editor of this column. Contact him at [email protected].

Obesity not only increases a patient’s lifetime risk of numerous chronic conditions, such as diabetes, heart disease and kidney disease, but it also is a major health issue during pregnancy. Women who are obese in pregnancy have a significantly higher chance of developing adverse perinatal outcomes and experiencing various complications that affect both their health and that of their babies.

With an ever-increasing population of overweight and obese women of reproductive age, as key caregivers for women, we must reexamine our approaches and do much more than microfocusing on a woman’s pre- and postdelivery health. We must play a more active role in helping our patients establish and maintain a healthy lifestyle – one that will help ward off and reduce the incidence of this concerning condition.

Over the previous two Master Class installments on obstetrics, we discussed the extent of the obesity epidemic and its link to diabetes, the alarming number of infants, children, and adolescents who are obese, and the implications of these societal and medical trends for ob.gyns.

In the July Master Class, we discussed the importance of appropriately counseling patients on healthy weight gain and physical activity in pregnancy. Because ob.gyns. may be the only health care professionals that many women may see, it is becoming more important that we help our patients and their children attain and maintain positive health and well-being.

In our September installment, Dr. Thomas R. Moore looked at obesity trends through the lens of the Barker Hypothesis, which got us thinking more than 3 decades ago about the role of intrauterine environment in short- and long-term health of offspring. Dr. Moore discussed how obesity in pregnancy appears to program offspring for downstream cardiovascular risk in adulthood.

He told us that we must not only liberally treat gestational diabetes and optimize glucose control during pregnancy, but, most importantly, we also must emphasize to women the importance of having healthy weights at the time of conception.

This month’s Master Class examines this latter concept in more depth. Dr. Patrick Catalano, professor in the department of obstetrics and gynecology and director of the Center for Reproductive Health at MetroHealth Medical Center, Case Western Reserve University, Cleveland, has been at the forefront of research on the physiologic impact of obesity on the placenta and the fetus, and on approaches for addressing maternal obesity and improving perinatal outcomes.

Dr. Catalano explains here why weight loss before pregnancy appears to be important for preventing adverse perinatal outcomes and breaking the intergenerational transfer of obesity.

Dr. Reece, who specializes in maternal-fetal medicine, is vice president for medical affairs at the University of Maryland, Baltimore, as well as the John Z. and Akiko K. Bowers Distinguished Professor and dean of the school of medicine. Dr. Reece said he had no relevant financial disclosures. He is the medical editor of this column. Contact him at [email protected].

Physeal separation of the distal humerus in a newborn is a rare and severe injury that requires immediate treatment. This fracture was reported as an extremely rare complication of cesarean section.¹ The correct diagnosis can be established by clinical and radiologic findings. However, this injury can be easily overlooked and misdiagnosed. Presentation often involves swelling, tenderness, and agitation with movement of the elbow.

We report a case in which neonatal physeal separation of the distal humerus occurred during cesarean section. The diagnosis was based on clinical and radiologic/arthrographic findings and treated with closed reduction and percutaneous fixation. The patient’s guardian provided written informed consent for print and electronic publication of this case report.

Case Report

A full-term (40-week gestation) male neonate weighing 3690 g was born through cesarean section at the mother’s request. Apgar score was 9 at 1 minute and 10 at 5 minutes. The vertex position of the fetus was confirmed with preoperative ultrasonography. This was the mother’s first pregnancy and an in vitro fertilization. On his second day of life, the patient was referred to the orthopedic department for evaluation of local swelling and diminished spontaneous motion of the right elbow.

Examination revealed local tenderness and swelling in the anterior and lateral aspects of the elbow. Passive elbow range of motion (ROM) caused agitation, and elbow instability was present. A complete neurovascular examination was performed, and neurovascular injury and compartment syndrome were ruled out. Hematologic workup showed no signs of septic arthritis. Radiographs showed posteromedial displacement of the humeroulnar joint. The patient was placed in a long-arm splint, and no reduction was attempted initially.

The patient was taken to the operating room the same day. With the patient under general anesthesia, an arthrogram of the right elbow was obtained. It showed posteromedial displacement of the distal humeral epiphysis (Figure 1A). Closed reduction was performed, and the quality of the reduction was confirmed by intraoperative imaging. Percutaneously, a single 2-mm Kirschner wire (K-wire) was placed in an oblique fashion from the inferolateral aspect of the distal fragment to the contralateral metaphysis of the humerus (Figure 1B). The patient was put in a long-arm splint with the elbow flexed at 90° and the forearm in midpronation.

Follow-up visits were scheduled for 1 week, 3 weeks, and 5 weeks after surgery. Three weeks after surgery, callus formation was confirmed, and the K-wire was removed. Five weeks after surgery, the long-arm splint was removed.

At 6-month follow-up, the patient was pain-free and had full elbow ROM, and radiographs (Figures 2A, 2B) confirmed anatomical restoration of the fracture.

Discussion

Madsen² reported the incidence of birth-related long-bone fractures, including fractures of the humerus, the femur, and the tibia (< 0.1%). According to that review, only 1 of 105,119 patients sustained traumatic physeal separation of the distal humerus.

Different mechanisms have been described for this rare fracture. As the physeal region is the weakest part of the distal humerus, it is prone to injury by rotational shear forces,^3,4 hyperextension of the elbow, or a backward thrust on the forearm with the elbow flexed.⁵ Excessive traction applied during cesarean delivery might cause physeal separation, which was the possible cause in the present case. Most patients have a complicated birth history.

This injury should be suspected in an irritable newborn with swelling, tenderness, and reduced mobility of the upper extremity. Osteomyelitis and septic arthritis should be considered in the differential diagnosis. Brachial plexus injury and dislocation of the elbow joint should also be kept in mind. Child abuse and metabolic bone diseases (eg, osteogenesis imperfecta) should also be considered.

Anteroposterior and lateral plain radiographs of the elbow usually establish the diagnosis. Alteration of humeroulnar alignment and displacement of the proximal forearm are the key points leading to the diagnosis.

The cartilaginous part of the distal humerus and humeroulnar alignment can be demonstrated by ultrasonography.⁶ Magnetic resonance imaging (MRI) can be helpful in diagnosis but is seldom required,⁷ and the sedation or general anesthesia used is a disadvantage. Arthrography is useful not only in diagnosis but in determining the quality of the reduction.⁸ An arthrogram may show that open reduction is unnecessary.

Treatment differs widely. In neonates, who have a tremendous healing capability, this fracture almost always heals uneventfully. An effective treatment method is closed reduction and cast immobilization. However, valgus malalignment and limited elbow ROM were noted in 5% of the patients treated with this method.⁴

Jacobsen and colleagues⁴ reported on 6 neonates who sustained traumatic separation of the distal epiphysis of the humerus at birth and who were treated with casting with or without closed reduction. The authors described good results. One patient had varus malalignment, which was attributed to fragment internal rotation caused by rotational instability.

As our patient’s instability was noted during surgery, we performed percutaneous pinning after arthrography-assisted closed reduction. We considered using 2 lateral pins for fixation, but, after the first pin was placed, fluoroscopic stress testing with the patient under anesthesia demonstrated adequate stability. A second, smaller pin could have been used to control rotation, if needed. Medial pin placement that avoids the ulnar nerve is difficult in the newborn elbow; medial pins should probably be avoided in the newborn, if possible.

Early diagnosis and treatment are essential. Late diagnosis was reported to lead to complications such as varus deformity and restriction of joint ROM.⁴

Our patient healed without any complications and achieved full ROM. Long-term follow-up is needed to diagnose any physeal bar that might lead to secondary deformities.

Conclusion

Cesarean section is reported to reduce birth complications, but it might cause fractures of the femur and humerus.¹ Avoiding application of excessive traction to the forearm can prevent physeal separation of the distal humerus. This entity should be kept in mind as a potential complication of cesarean section. Arthrography is helpful in treatment and may help avoid unnecessary open reduction.

Physeal separation of the distal humerus in a newborn is a rare and severe injury that requires immediate treatment. This fracture was reported as an extremely rare complication of cesarean section.¹ The correct diagnosis can be established by clinical and radiologic findings. However, this injury can be easily overlooked and misdiagnosed. Presentation often involves swelling, tenderness, and agitation with movement of the elbow.

We report a case in which neonatal physeal separation of the distal humerus occurred during cesarean section. The diagnosis was based on clinical and radiologic/arthrographic findings and treated with closed reduction and percutaneous fixation. The patient’s guardian provided written informed consent for print and electronic publication of this case report.

Case Report

A full-term (40-week gestation) male neonate weighing 3690 g was born through cesarean section at the mother’s request. Apgar score was 9 at 1 minute and 10 at 5 minutes. The vertex position of the fetus was confirmed with preoperative ultrasonography. This was the mother’s first pregnancy and an in vitro fertilization. On his second day of life, the patient was referred to the orthopedic department for evaluation of local swelling and diminished spontaneous motion of the right elbow.

Examination revealed local tenderness and swelling in the anterior and lateral aspects of the elbow. Passive elbow range of motion (ROM) caused agitation, and elbow instability was present. A complete neurovascular examination was performed, and neurovascular injury and compartment syndrome were ruled out. Hematologic workup showed no signs of septic arthritis. Radiographs showed posteromedial displacement of the humeroulnar joint. The patient was placed in a long-arm splint, and no reduction was attempted initially.

The patient was taken to the operating room the same day. With the patient under general anesthesia, an arthrogram of the right elbow was obtained. It showed posteromedial displacement of the distal humeral epiphysis (Figure 1A). Closed reduction was performed, and the quality of the reduction was confirmed by intraoperative imaging. Percutaneously, a single 2-mm Kirschner wire (K-wire) was placed in an oblique fashion from the inferolateral aspect of the distal fragment to the contralateral metaphysis of the humerus (Figure 1B). The patient was put in a long-arm splint with the elbow flexed at 90° and the forearm in midpronation.

Follow-up visits were scheduled for 1 week, 3 weeks, and 5 weeks after surgery. Three weeks after surgery, callus formation was confirmed, and the K-wire was removed. Five weeks after surgery, the long-arm splint was removed.

At 6-month follow-up, the patient was pain-free and had full elbow ROM, and radiographs (Figures 2A, 2B) confirmed anatomical restoration of the fracture.

Discussion

Madsen² reported the incidence of birth-related long-bone fractures, including fractures of the humerus, the femur, and the tibia (< 0.1%). According to that review, only 1 of 105,119 patients sustained traumatic physeal separation of the distal humerus.

Different mechanisms have been described for this rare fracture. As the physeal region is the weakest part of the distal humerus, it is prone to injury by rotational shear forces,^3,4 hyperextension of the elbow, or a backward thrust on the forearm with the elbow flexed.⁵ Excessive traction applied during cesarean delivery might cause physeal separation, which was the possible cause in the present case. Most patients have a complicated birth history.

This injury should be suspected in an irritable newborn with swelling, tenderness, and reduced mobility of the upper extremity. Osteomyelitis and septic arthritis should be considered in the differential diagnosis. Brachial plexus injury and dislocation of the elbow joint should also be kept in mind. Child abuse and metabolic bone diseases (eg, osteogenesis imperfecta) should also be considered.

Anteroposterior and lateral plain radiographs of the elbow usually establish the diagnosis. Alteration of humeroulnar alignment and displacement of the proximal forearm are the key points leading to the diagnosis.

The cartilaginous part of the distal humerus and humeroulnar alignment can be demonstrated by ultrasonography.⁶ Magnetic resonance imaging (MRI) can be helpful in diagnosis but is seldom required,⁷ and the sedation or general anesthesia used is a disadvantage. Arthrography is useful not only in diagnosis but in determining the quality of the reduction.⁸ An arthrogram may show that open reduction is unnecessary.

Treatment differs widely. In neonates, who have a tremendous healing capability, this fracture almost always heals uneventfully. An effective treatment method is closed reduction and cast immobilization. However, valgus malalignment and limited elbow ROM were noted in 5% of the patients treated with this method.⁴

Jacobsen and colleagues⁴ reported on 6 neonates who sustained traumatic separation of the distal epiphysis of the humerus at birth and who were treated with casting with or without closed reduction. The authors described good results. One patient had varus malalignment, which was attributed to fragment internal rotation caused by rotational instability.

As our patient’s instability was noted during surgery, we performed percutaneous pinning after arthrography-assisted closed reduction. We considered using 2 lateral pins for fixation, but, after the first pin was placed, fluoroscopic stress testing with the patient under anesthesia demonstrated adequate stability. A second, smaller pin could have been used to control rotation, if needed. Medial pin placement that avoids the ulnar nerve is difficult in the newborn elbow; medial pins should probably be avoided in the newborn, if possible.

Early diagnosis and treatment are essential. Late diagnosis was reported to lead to complications such as varus deformity and restriction of joint ROM.⁴

Our patient healed without any complications and achieved full ROM. Long-term follow-up is needed to diagnose any physeal bar that might lead to secondary deformities.

Conclusion

Cesarean section is reported to reduce birth complications, but it might cause fractures of the femur and humerus.¹ Avoiding application of excessive traction to the forearm can prevent physeal separation of the distal humerus. This entity should be kept in mind as a potential complication of cesarean section. Arthrography is helpful in treatment and may help avoid unnecessary open reduction.

Physeal separation of the distal humerus in a newborn is a rare and severe injury that requires immediate treatment. This fracture was reported as an extremely rare complication of cesarean section.¹ The correct diagnosis can be established by clinical and radiologic findings. However, this injury can be easily overlooked and misdiagnosed. Presentation often involves swelling, tenderness, and agitation with movement of the elbow.

We report a case in which neonatal physeal separation of the distal humerus occurred during cesarean section. The diagnosis was based on clinical and radiologic/arthrographic findings and treated with closed reduction and percutaneous fixation. The patient’s guardian provided written informed consent for print and electronic publication of this case report.

Case Report

A full-term (40-week gestation) male neonate weighing 3690 g was born through cesarean section at the mother’s request. Apgar score was 9 at 1 minute and 10 at 5 minutes. The vertex position of the fetus was confirmed with preoperative ultrasonography. This was the mother’s first pregnancy and an in vitro fertilization. On his second day of life, the patient was referred to the orthopedic department for evaluation of local swelling and diminished spontaneous motion of the right elbow.

Examination revealed local tenderness and swelling in the anterior and lateral aspects of the elbow. Passive elbow range of motion (ROM) caused agitation, and elbow instability was present. A complete neurovascular examination was performed, and neurovascular injury and compartment syndrome were ruled out. Hematologic workup showed no signs of septic arthritis. Radiographs showed posteromedial displacement of the humeroulnar joint. The patient was placed in a long-arm splint, and no reduction was attempted initially.

The patient was taken to the operating room the same day. With the patient under general anesthesia, an arthrogram of the right elbow was obtained. It showed posteromedial displacement of the distal humeral epiphysis (Figure 1A). Closed reduction was performed, and the quality of the reduction was confirmed by intraoperative imaging. Percutaneously, a single 2-mm Kirschner wire (K-wire) was placed in an oblique fashion from the inferolateral aspect of the distal fragment to the contralateral metaphysis of the humerus (Figure 1B). The patient was put in a long-arm splint with the elbow flexed at 90° and the forearm in midpronation.

Follow-up visits were scheduled for 1 week, 3 weeks, and 5 weeks after surgery. Three weeks after surgery, callus formation was confirmed, and the K-wire was removed. Five weeks after surgery, the long-arm splint was removed.

At 6-month follow-up, the patient was pain-free and had full elbow ROM, and radiographs (Figures 2A, 2B) confirmed anatomical restoration of the fracture.

Discussion

Madsen² reported the incidence of birth-related long-bone fractures, including fractures of the humerus, the femur, and the tibia (< 0.1%). According to that review, only 1 of 105,119 patients sustained traumatic physeal separation of the distal humerus.

Different mechanisms have been described for this rare fracture. As the physeal region is the weakest part of the distal humerus, it is prone to injury by rotational shear forces,^3,4 hyperextension of the elbow, or a backward thrust on the forearm with the elbow flexed.⁵ Excessive traction applied during cesarean delivery might cause physeal separation, which was the possible cause in the present case. Most patients have a complicated birth history.

This injury should be suspected in an irritable newborn with swelling, tenderness, and reduced mobility of the upper extremity. Osteomyelitis and septic arthritis should be considered in the differential diagnosis. Brachial plexus injury and dislocation of the elbow joint should also be kept in mind. Child abuse and metabolic bone diseases (eg, osteogenesis imperfecta) should also be considered.

Anteroposterior and lateral plain radiographs of the elbow usually establish the diagnosis. Alteration of humeroulnar alignment and displacement of the proximal forearm are the key points leading to the diagnosis.

The cartilaginous part of the distal humerus and humeroulnar alignment can be demonstrated by ultrasonography.⁶ Magnetic resonance imaging (MRI) can be helpful in diagnosis but is seldom required,⁷ and the sedation or general anesthesia used is a disadvantage. Arthrography is useful not only in diagnosis but in determining the quality of the reduction.⁸ An arthrogram may show that open reduction is unnecessary.

Treatment differs widely. In neonates, who have a tremendous healing capability, this fracture almost always heals uneventfully. An effective treatment method is closed reduction and cast immobilization. However, valgus malalignment and limited elbow ROM were noted in 5% of the patients treated with this method.⁴

Jacobsen and colleagues⁴ reported on 6 neonates who sustained traumatic separation of the distal epiphysis of the humerus at birth and who were treated with casting with or without closed reduction. The authors described good results. One patient had varus malalignment, which was attributed to fragment internal rotation caused by rotational instability.

As our patient’s instability was noted during surgery, we performed percutaneous pinning after arthrography-assisted closed reduction. We considered using 2 lateral pins for fixation, but, after the first pin was placed, fluoroscopic stress testing with the patient under anesthesia demonstrated adequate stability. A second, smaller pin could have been used to control rotation, if needed. Medial pin placement that avoids the ulnar nerve is difficult in the newborn elbow; medial pins should probably be avoided in the newborn, if possible.

Early diagnosis and treatment are essential. Late diagnosis was reported to lead to complications such as varus deformity and restriction of joint ROM.⁴

Our patient healed without any complications and achieved full ROM. Long-term follow-up is needed to diagnose any physeal bar that might lead to secondary deformities.

Conclusion

Cesarean section is reported to reduce birth complications, but it might cause fractures of the femur and humerus.¹ Avoiding application of excessive traction to the forearm can prevent physeal separation of the distal humerus. This entity should be kept in mind as a potential complication of cesarean section. Arthrography is helpful in treatment and may help avoid unnecessary open reduction.

Chordomas persist as one of the rarer malignancies, accounting for approximately 1% to 4% of primary bone cancers.¹ When chordomas occur, these tumors localize predominantly in the sacrococcygeal region.² In addition to the urgency for addressing a relatively fast-growing tumor, the anatomical complexity of this area complicates the potential treatments. Furthermore, because of the lack of definitive symptoms, diagnosis is often difficult and typically occurs later in the disease progression.³ An aggressive treatment approach is often warranted because of the biologically aggressive nature of this disease. Full or partial sacrectomy is often the only option that offers the possibility of a long-term cure.⁴ A sacrectomy is a destructive procedure that can lead to mechanical instability depending on the extent of the surgical resection. When the entire sacrum is removed, there is an obvious need for lumbar-pelvic fixation; however, traditionally, partial sacrectomy procedures have been successfully performed without the need for instrumentation.^3,4

This report describes the case of a patient with a noninstrumented sacrectomy procedure distal to the S2 foramen that resulted in an insufficiency fracture. The patient provided written informed consent for print and electronic publication of this case report.

Case Report

A 66-year-old woman presented with severe lower back pain of a month’s duration. Her pain was localized to the coccyx area and did not radiate to the lower legs. Although the pain could not be elicited by palpation, pain occurred when sitting and increased when standing for prolonged periods. Three weeks prior to the patient’s initial office visit, she noticed transient constipation and urinary retention. She denied any fever, chills, nausea, vomiting, unexplained weight loss, weight gain, and abdominal pain. There were no motor deficits in the lower limbs. Sensation was intact in the lower limbs except for the posterior aspect of the left leg down to the popliteal fossa, where light touch perception was absent. She recalled the loss of sensation in this area 20 years earlier, and it had neither progressed nor abated since then. She had a history of osteoarthritis and had been diagnosed with degenerative disc disease 20 years ago.

A radiographic review of her lumbar spine showed significant spinal stenosis and degenerative disease of the lumbar spine on non–contrast-enhanced magnetic resonance imaging (MRI). The MRI also revealed a large, soft-tissue mass at the S3-S4 level, eroding most of the S3 vertebral body and extending into the S4 vertebral body. The MRI images used for this analysis were insufficient in providing a complete portrayal of the entire mass. Because of these uncertainties, contrast-enhanced and non–contrast-enhanced pelvic MRIs were taken. The MRI analyses identified a mass density replacing the lower sacrum and upper coccyx that was bright in intensity on T2 and dim on T1 sequences. Sagittal imaging measurements were 5.9×2.5 cm and 4.4 cm right-to-left on coronal imaging. The mass extended beyond the involved sacrococcygeal segments and dorsally beyond the normal cortical margin of the sacrum and coccyx (Figures 1A, 1B). Next, a computer tomographic–guided needle biopsy through a posterior paraspinal approach was obtained. The biopsy consisted of fragments of a malignant neoplasm consistent with physaliferous cells. The specimen was positive for pankeratin, keratin AE1/AE3, epithelial membrane antigen, and S100 protein. This supported a diagnosis of a sacral chordoma. An en bloc sacrectomy at S2; lumbar laminectomy at L5, S1, and S2; and thecal sac transection at the S3 nerve roots were planned.

Surgical Procedure

The patient was placed in the prone position after a colostomy and harvesting of a rectus flap in the supine position. A midline incision was made from the spinous process of L5 down through the tip of the coccyx, and soft tissues were elevated while maintaining hemostasis. The most distal part of the coccyx was transected, and using a combination of electrocautery and paraspinal elevators, rectal and peritoneal tissues were elevated off the ventral component of the coccyx until a hand could easily reach the bifurcation of the iliac vessels. Electrocautery transected paraspinal muscles at the S1 and S2 levels while the more cranial paraspinal musculature was elevated to allow for a laminectomy. The spinous processes were removed from L5 and the sacrum with a Leksell rongeur. A high-speed burr thinned the dorsal lamina components of L5, S1, and the leading edge of S2. The L5, S1, and S2 nerve roots were identified. The gluteal muscles were elevated and the sacral coccygeal ligaments were transected. After identifying the sciatic notches, the S2 nerves exiting the foramen were identified, followed out through the sciatic notch, and a wire was passed through this region. Three 2-0 silk ties were applied to the exposed portion of the S3 and S4 nerve roots, and the nerves were transected because they were integrally involved with the tumor. Using a series of high-speed burrs and osteotomes, lateral cuts were made through the sciatic notch. The sacrum was osteotomized at the S2 sacral foramen through the anterior component with an osteotome, while a hand protected the ventral structures. The remaining parts of the S3 and S4 dorsal nerve roots were transected. An incision through the peritoneum was made to access the rectus flap, and a plastic surgeon closed the wounds and secured the flap. 

Postoperative Course

The patient’s final pathology confirmed a chordoma with negative margins. Postoperatively, the rectus flap became ischemic and a wound infection developed. It was irrigated, débrided, and treated with vacuum-assisted closure (VAC), in addition to perioperative antibiotic administration. An abdominal computed tomography (CT) scan did not show any fistula, and her wound remained healthy, pink, and viable as her VAC was changed every 3 days. Because the patient’s nutritional status was compromised, she started nutritional supplements in addition to a regular diet. Physical therapy was prescribed and the patient began bladder training with self-catheterization after a failed voiding trial attempt. After 2 months of convalescence, the patient had mobilized well and had progressed to walking without an ambulatory aide.

At her third postoperative month, the patient noted new onset of extreme pain in the groin and left thigh regions. The patient was examined and appeared to have a stable neurological exam. She had reproducible pain with a FABER (Flexion, Abduction, External Rotation, and Extension) test. MRI showed increased signal on short tau inversion recovery (STIR) sequences and T2-weighted images that was consistent with a left sacral ala stress fracture with a vertically oriented fracture line (Figures 2A, 2B). The patient was asked to begin utilizing a walker for ambulatory assistance, but her weight-bearing status was not changed. Over the course of 3 months, the patient noted a resolution of her pain. All postoperative MRI images confirmed the patient to be disease-free; and in addition, all of her follow-up radiographs showed a stable pelvic ring (Figures 3A, 3B). At her 2-year follow-up, the patient remained disease- and pain-free.

Discussion

Full discussions of the mechanical considerations of a partial sacrectomy have been described previously^5-8; however, surgeons typically consider the need for lumbar-pelvic stabilization when the surgical resection requires a violation of the S1 body. Approximately two-thirds of sacral tumors occur at or below the level of the S2 body.⁸ These lesions of the caudal sacrum can sometimes be effectively resected with transverse partial sacrectomy. Great care is taken to resect only the portion of the sacrum necessary for local disease control, sparing as much of the sacroiliac joint and as many of the lumbosacral nerve roots as possible.

Under normal conditions, the sacroiliac articulation is stabilized by both its geometric interface and its extraordinarily strong ligaments. This spatial arrangement conveys stability primarily against caudal migration of the sacrum. The sacroiliac, sacrotuberous, sacrospinous, and lumbosacral ligaments, which are among the strongest ligaments in the body, primarily act to provide stability to the pelvic ring by preventing diastasis. The combination of these factors renders the spinopelvic segment especially stable. Previously, 2 biomechanical studies that specifically looked at extreme loading patterns to better understand the need for lumbar-pelvic instrumentation predicted a fracture pattern when there was an inability of the base of the sacral ala to resist shear.^8,9 This is precisely where our patient’s insufficiency fracture occurred.

To our knowledge, this is the first reported in vivo evidence of this fracture pattern. While this patient’s potential history of osteoporosis may have elevated or contributed to her risk for fracture, her preoperative bone densitometry, with T scores of -1.0 on the left and right femur necks and 0.8 on her L1-L4 anteroposterior spine, would argue against this risk factor. None of these values represent a truly osteoporotic patient. It would appear that our patient sustained the fracture pattern predicted by Hugate and colleagues.⁸

The edema seen on the MRI most likely represents a fracture; however, sacroiliitis and infection are also potential diagnoses. Because there was no tumor in this region on the preoperative scans, we thought that a residual tumor was unlikely. The signal changes seen on T2 MRI sequences represent edema. The use of a bone scan that detects healing bone may have been a useful additional study to confirm this fracture as opposed to sacroiliitis. A CT scan would have been a potentially useful study to provide detail of fracture displacement and the overall fracture pattern. Standing plain radiographs are best for viewing fracture displacement with weight-bearing.

Surgeons contemplating performing partial sacrectomies should bear in mind that, even with preservation of the S1 body, a potential for fracture exists as evidenced by our patient. In our opinion, this patient did not require instrumentation but a more gradual rehabilitation program.

Chordomas persist as one of the rarer malignancies, accounting for approximately 1% to 4% of primary bone cancers.¹ When chordomas occur, these tumors localize predominantly in the sacrococcygeal region.² In addition to the urgency for addressing a relatively fast-growing tumor, the anatomical complexity of this area complicates the potential treatments. Furthermore, because of the lack of definitive symptoms, diagnosis is often difficult and typically occurs later in the disease progression.³ An aggressive treatment approach is often warranted because of the biologically aggressive nature of this disease. Full or partial sacrectomy is often the only option that offers the possibility of a long-term cure.⁴ A sacrectomy is a destructive procedure that can lead to mechanical instability depending on the extent of the surgical resection. When the entire sacrum is removed, there is an obvious need for lumbar-pelvic fixation; however, traditionally, partial sacrectomy procedures have been successfully performed without the need for instrumentation.^3,4

This report describes the case of a patient with a noninstrumented sacrectomy procedure distal to the S2 foramen that resulted in an insufficiency fracture. The patient provided written informed consent for print and electronic publication of this case report.

Case Report

A 66-year-old woman presented with severe lower back pain of a month’s duration. Her pain was localized to the coccyx area and did not radiate to the lower legs. Although the pain could not be elicited by palpation, pain occurred when sitting and increased when standing for prolonged periods. Three weeks prior to the patient’s initial office visit, she noticed transient constipation and urinary retention. She denied any fever, chills, nausea, vomiting, unexplained weight loss, weight gain, and abdominal pain. There were no motor deficits in the lower limbs. Sensation was intact in the lower limbs except for the posterior aspect of the left leg down to the popliteal fossa, where light touch perception was absent. She recalled the loss of sensation in this area 20 years earlier, and it had neither progressed nor abated since then. She had a history of osteoarthritis and had been diagnosed with degenerative disc disease 20 years ago.

A radiographic review of her lumbar spine showed significant spinal stenosis and degenerative disease of the lumbar spine on non–contrast-enhanced magnetic resonance imaging (MRI). The MRI also revealed a large, soft-tissue mass at the S3-S4 level, eroding most of the S3 vertebral body and extending into the S4 vertebral body. The MRI images used for this analysis were insufficient in providing a complete portrayal of the entire mass. Because of these uncertainties, contrast-enhanced and non–contrast-enhanced pelvic MRIs were taken. The MRI analyses identified a mass density replacing the lower sacrum and upper coccyx that was bright in intensity on T2 and dim on T1 sequences. Sagittal imaging measurements were 5.9×2.5 cm and 4.4 cm right-to-left on coronal imaging. The mass extended beyond the involved sacrococcygeal segments and dorsally beyond the normal cortical margin of the sacrum and coccyx (Figures 1A, 1B). Next, a computer tomographic–guided needle biopsy through a posterior paraspinal approach was obtained. The biopsy consisted of fragments of a malignant neoplasm consistent with physaliferous cells. The specimen was positive for pankeratin, keratin AE1/AE3, epithelial membrane antigen, and S100 protein. This supported a diagnosis of a sacral chordoma. An en bloc sacrectomy at S2; lumbar laminectomy at L5, S1, and S2; and thecal sac transection at the S3 nerve roots were planned.

Surgical Procedure

The patient was placed in the prone position after a colostomy and harvesting of a rectus flap in the supine position. A midline incision was made from the spinous process of L5 down through the tip of the coccyx, and soft tissues were elevated while maintaining hemostasis. The most distal part of the coccyx was transected, and using a combination of electrocautery and paraspinal elevators, rectal and peritoneal tissues were elevated off the ventral component of the coccyx until a hand could easily reach the bifurcation of the iliac vessels. Electrocautery transected paraspinal muscles at the S1 and S2 levels while the more cranial paraspinal musculature was elevated to allow for a laminectomy. The spinous processes were removed from L5 and the sacrum with a Leksell rongeur. A high-speed burr thinned the dorsal lamina components of L5, S1, and the leading edge of S2. The L5, S1, and S2 nerve roots were identified. The gluteal muscles were elevated and the sacral coccygeal ligaments were transected. After identifying the sciatic notches, the S2 nerves exiting the foramen were identified, followed out through the sciatic notch, and a wire was passed through this region. Three 2-0 silk ties were applied to the exposed portion of the S3 and S4 nerve roots, and the nerves were transected because they were integrally involved with the tumor. Using a series of high-speed burrs and osteotomes, lateral cuts were made through the sciatic notch. The sacrum was osteotomized at the S2 sacral foramen through the anterior component with an osteotome, while a hand protected the ventral structures. The remaining parts of the S3 and S4 dorsal nerve roots were transected. An incision through the peritoneum was made to access the rectus flap, and a plastic surgeon closed the wounds and secured the flap. 

Postoperative Course

The patient’s final pathology confirmed a chordoma with negative margins. Postoperatively, the rectus flap became ischemic and a wound infection developed. It was irrigated, débrided, and treated with vacuum-assisted closure (VAC), in addition to perioperative antibiotic administration. An abdominal computed tomography (CT) scan did not show any fistula, and her wound remained healthy, pink, and viable as her VAC was changed every 3 days. Because the patient’s nutritional status was compromised, she started nutritional supplements in addition to a regular diet. Physical therapy was prescribed and the patient began bladder training with self-catheterization after a failed voiding trial attempt. After 2 months of convalescence, the patient had mobilized well and had progressed to walking without an ambulatory aide.

At her third postoperative month, the patient noted new onset of extreme pain in the groin and left thigh regions. The patient was examined and appeared to have a stable neurological exam. She had reproducible pain with a FABER (Flexion, Abduction, External Rotation, and Extension) test. MRI showed increased signal on short tau inversion recovery (STIR) sequences and T2-weighted images that was consistent with a left sacral ala stress fracture with a vertically oriented fracture line (Figures 2A, 2B). The patient was asked to begin utilizing a walker for ambulatory assistance, but her weight-bearing status was not changed. Over the course of 3 months, the patient noted a resolution of her pain. All postoperative MRI images confirmed the patient to be disease-free; and in addition, all of her follow-up radiographs showed a stable pelvic ring (Figures 3A, 3B). At her 2-year follow-up, the patient remained disease- and pain-free.

Discussion

Full discussions of the mechanical considerations of a partial sacrectomy have been described previously^5-8; however, surgeons typically consider the need for lumbar-pelvic stabilization when the surgical resection requires a violation of the S1 body. Approximately two-thirds of sacral tumors occur at or below the level of the S2 body.⁸ These lesions of the caudal sacrum can sometimes be effectively resected with transverse partial sacrectomy. Great care is taken to resect only the portion of the sacrum necessary for local disease control, sparing as much of the sacroiliac joint and as many of the lumbosacral nerve roots as possible.

Under normal conditions, the sacroiliac articulation is stabilized by both its geometric interface and its extraordinarily strong ligaments. This spatial arrangement conveys stability primarily against caudal migration of the sacrum. The sacroiliac, sacrotuberous, sacrospinous, and lumbosacral ligaments, which are among the strongest ligaments in the body, primarily act to provide stability to the pelvic ring by preventing diastasis. The combination of these factors renders the spinopelvic segment especially stable. Previously, 2 biomechanical studies that specifically looked at extreme loading patterns to better understand the need for lumbar-pelvic instrumentation predicted a fracture pattern when there was an inability of the base of the sacral ala to resist shear.^8,9 This is precisely where our patient’s insufficiency fracture occurred.

To our knowledge, this is the first reported in vivo evidence of this fracture pattern. While this patient’s potential history of osteoporosis may have elevated or contributed to her risk for fracture, her preoperative bone densitometry, with T scores of -1.0 on the left and right femur necks and 0.8 on her L1-L4 anteroposterior spine, would argue against this risk factor. None of these values represent a truly osteoporotic patient. It would appear that our patient sustained the fracture pattern predicted by Hugate and colleagues.⁸

The edema seen on the MRI most likely represents a fracture; however, sacroiliitis and infection are also potential diagnoses. Because there was no tumor in this region on the preoperative scans, we thought that a residual tumor was unlikely. The signal changes seen on T2 MRI sequences represent edema. The use of a bone scan that detects healing bone may have been a useful additional study to confirm this fracture as opposed to sacroiliitis. A CT scan would have been a potentially useful study to provide detail of fracture displacement and the overall fracture pattern. Standing plain radiographs are best for viewing fracture displacement with weight-bearing.

Surgeons contemplating performing partial sacrectomies should bear in mind that, even with preservation of the S1 body, a potential for fracture exists as evidenced by our patient. In our opinion, this patient did not require instrumentation but a more gradual rehabilitation program.

Chordomas persist as one of the rarer malignancies, accounting for approximately 1% to 4% of primary bone cancers.¹ When chordomas occur, these tumors localize predominantly in the sacrococcygeal region.² In addition to the urgency for addressing a relatively fast-growing tumor, the anatomical complexity of this area complicates the potential treatments. Furthermore, because of the lack of definitive symptoms, diagnosis is often difficult and typically occurs later in the disease progression.³ An aggressive treatment approach is often warranted because of the biologically aggressive nature of this disease. Full or partial sacrectomy is often the only option that offers the possibility of a long-term cure.⁴ A sacrectomy is a destructive procedure that can lead to mechanical instability depending on the extent of the surgical resection. When the entire sacrum is removed, there is an obvious need for lumbar-pelvic fixation; however, traditionally, partial sacrectomy procedures have been successfully performed without the need for instrumentation.^3,4

This report describes the case of a patient with a noninstrumented sacrectomy procedure distal to the S2 foramen that resulted in an insufficiency fracture. The patient provided written informed consent for print and electronic publication of this case report.

Case Report

A 66-year-old woman presented with severe lower back pain of a month’s duration. Her pain was localized to the coccyx area and did not radiate to the lower legs. Although the pain could not be elicited by palpation, pain occurred when sitting and increased when standing for prolonged periods. Three weeks prior to the patient’s initial office visit, she noticed transient constipation and urinary retention. She denied any fever, chills, nausea, vomiting, unexplained weight loss, weight gain, and abdominal pain. There were no motor deficits in the lower limbs. Sensation was intact in the lower limbs except for the posterior aspect of the left leg down to the popliteal fossa, where light touch perception was absent. She recalled the loss of sensation in this area 20 years earlier, and it had neither progressed nor abated since then. She had a history of osteoarthritis and had been diagnosed with degenerative disc disease 20 years ago.

A radiographic review of her lumbar spine showed significant spinal stenosis and degenerative disease of the lumbar spine on non–contrast-enhanced magnetic resonance imaging (MRI). The MRI also revealed a large, soft-tissue mass at the S3-S4 level, eroding most of the S3 vertebral body and extending into the S4 vertebral body. The MRI images used for this analysis were insufficient in providing a complete portrayal of the entire mass. Because of these uncertainties, contrast-enhanced and non–contrast-enhanced pelvic MRIs were taken. The MRI analyses identified a mass density replacing the lower sacrum and upper coccyx that was bright in intensity on T2 and dim on T1 sequences. Sagittal imaging measurements were 5.9×2.5 cm and 4.4 cm right-to-left on coronal imaging. The mass extended beyond the involved sacrococcygeal segments and dorsally beyond the normal cortical margin of the sacrum and coccyx (Figures 1A, 1B). Next, a computer tomographic–guided needle biopsy through a posterior paraspinal approach was obtained. The biopsy consisted of fragments of a malignant neoplasm consistent with physaliferous cells. The specimen was positive for pankeratin, keratin AE1/AE3, epithelial membrane antigen, and S100 protein. This supported a diagnosis of a sacral chordoma. An en bloc sacrectomy at S2; lumbar laminectomy at L5, S1, and S2; and thecal sac transection at the S3 nerve roots were planned.

Surgical Procedure

The patient was placed in the prone position after a colostomy and harvesting of a rectus flap in the supine position. A midline incision was made from the spinous process of L5 down through the tip of the coccyx, and soft tissues were elevated while maintaining hemostasis. The most distal part of the coccyx was transected, and using a combination of electrocautery and paraspinal elevators, rectal and peritoneal tissues were elevated off the ventral component of the coccyx until a hand could easily reach the bifurcation of the iliac vessels. Electrocautery transected paraspinal muscles at the S1 and S2 levels while the more cranial paraspinal musculature was elevated to allow for a laminectomy. The spinous processes were removed from L5 and the sacrum with a Leksell rongeur. A high-speed burr thinned the dorsal lamina components of L5, S1, and the leading edge of S2. The L5, S1, and S2 nerve roots were identified. The gluteal muscles were elevated and the sacral coccygeal ligaments were transected. After identifying the sciatic notches, the S2 nerves exiting the foramen were identified, followed out through the sciatic notch, and a wire was passed through this region. Three 2-0 silk ties were applied to the exposed portion of the S3 and S4 nerve roots, and the nerves were transected because they were integrally involved with the tumor. Using a series of high-speed burrs and osteotomes, lateral cuts were made through the sciatic notch. The sacrum was osteotomized at the S2 sacral foramen through the anterior component with an osteotome, while a hand protected the ventral structures. The remaining parts of the S3 and S4 dorsal nerve roots were transected. An incision through the peritoneum was made to access the rectus flap, and a plastic surgeon closed the wounds and secured the flap. 

Postoperative Course

The patient’s final pathology confirmed a chordoma with negative margins. Postoperatively, the rectus flap became ischemic and a wound infection developed. It was irrigated, débrided, and treated with vacuum-assisted closure (VAC), in addition to perioperative antibiotic administration. An abdominal computed tomography (CT) scan did not show any fistula, and her wound remained healthy, pink, and viable as her VAC was changed every 3 days. Because the patient’s nutritional status was compromised, she started nutritional supplements in addition to a regular diet. Physical therapy was prescribed and the patient began bladder training with self-catheterization after a failed voiding trial attempt. After 2 months of convalescence, the patient had mobilized well and had progressed to walking without an ambulatory aide.

At her third postoperative month, the patient noted new onset of extreme pain in the groin and left thigh regions. The patient was examined and appeared to have a stable neurological exam. She had reproducible pain with a FABER (Flexion, Abduction, External Rotation, and Extension) test. MRI showed increased signal on short tau inversion recovery (STIR) sequences and T2-weighted images that was consistent with a left sacral ala stress fracture with a vertically oriented fracture line (Figures 2A, 2B). The patient was asked to begin utilizing a walker for ambulatory assistance, but her weight-bearing status was not changed. Over the course of 3 months, the patient noted a resolution of her pain. All postoperative MRI images confirmed the patient to be disease-free; and in addition, all of her follow-up radiographs showed a stable pelvic ring (Figures 3A, 3B). At her 2-year follow-up, the patient remained disease- and pain-free.

Discussion

Full discussions of the mechanical considerations of a partial sacrectomy have been described previously^5-8; however, surgeons typically consider the need for lumbar-pelvic stabilization when the surgical resection requires a violation of the S1 body. Approximately two-thirds of sacral tumors occur at or below the level of the S2 body.⁸ These lesions of the caudal sacrum can sometimes be effectively resected with transverse partial sacrectomy. Great care is taken to resect only the portion of the sacrum necessary for local disease control, sparing as much of the sacroiliac joint and as many of the lumbosacral nerve roots as possible.

Under normal conditions, the sacroiliac articulation is stabilized by both its geometric interface and its extraordinarily strong ligaments. This spatial arrangement conveys stability primarily against caudal migration of the sacrum. The sacroiliac, sacrotuberous, sacrospinous, and lumbosacral ligaments, which are among the strongest ligaments in the body, primarily act to provide stability to the pelvic ring by preventing diastasis. The combination of these factors renders the spinopelvic segment especially stable. Previously, 2 biomechanical studies that specifically looked at extreme loading patterns to better understand the need for lumbar-pelvic instrumentation predicted a fracture pattern when there was an inability of the base of the sacral ala to resist shear.^8,9 This is precisely where our patient’s insufficiency fracture occurred.

To our knowledge, this is the first reported in vivo evidence of this fracture pattern. While this patient’s potential history of osteoporosis may have elevated or contributed to her risk for fracture, her preoperative bone densitometry, with T scores of -1.0 on the left and right femur necks and 0.8 on her L1-L4 anteroposterior spine, would argue against this risk factor. None of these values represent a truly osteoporotic patient. It would appear that our patient sustained the fracture pattern predicted by Hugate and colleagues.⁸

The edema seen on the MRI most likely represents a fracture; however, sacroiliitis and infection are also potential diagnoses. Because there was no tumor in this region on the preoperative scans, we thought that a residual tumor was unlikely. The signal changes seen on T2 MRI sequences represent edema. The use of a bone scan that detects healing bone may have been a useful additional study to confirm this fracture as opposed to sacroiliitis. A CT scan would have been a potentially useful study to provide detail of fracture displacement and the overall fracture pattern. Standing plain radiographs are best for viewing fracture displacement with weight-bearing.

Surgeons contemplating performing partial sacrectomies should bear in mind that, even with preservation of the S1 body, a potential for fracture exists as evidenced by our patient. In our opinion, this patient did not require instrumentation but a more gradual rehabilitation program.

Blastomycosis is a rare fungal infection that primarily produces acute lung infections but may on occasion disseminate to multiple sites, including the skin, bone, central nervous system (CNS), and oropharynx.^1-30 In the case of a primary infection of the lung, if there is a high index of suspicion and a thorough diagnostic workup, the diagnosis can be made from sputum or bronchoscopy.²⁴ Patients present with acute pneumonia that either resolves spontaneously or proceeds to chronic pneumonia with extrapulmonary spread to multiple organs, including the spine. Once vertebral involvement occurs, an untreated infection may result in vertebral body destruction and paraspinal and epidural abscess formation followed by neurologic injury and loss of structural integrity of the spine.^{11,13,17,23,27,29}

In this article, we present a case of blastomycosis of the vertebral body and provide a detailed review of the literature concerning this extremely rare infection of the spine. The patient provided written informed consent for print and electronic publication of this case report.

Case Report

A 30-year-old African American man with known pulmonary blastomycosis, for which he had been treated with oral itraconazole 200 mg twice daily for 6 months, was admitted to the hospital with a 2-month history of mild thoracolumbar back pain. He reported transient numbness and tingling in the lower extremities but no weakness. He denied weight loss, fatigue, appetite loss, and significant night pain. On physical examination, he was alert and oriented, well nourished, and in no acute distress. Percussion revealed limited range of motion and pain. Further examination of the spine demonstrated no spasm, swelling, erythema, or drainage. The lower extremities had intact sensation, motor strength, reflexes, and pulses, and clonus was absent. White blood cell count was 8100 cells/μL (normal), erythrocyte sedimentation rate was 77 mm/h (normal range, 0-20 mm/h), and C-reactive protein level was 57.2 mg/L (normal, ≤ 10 mg/L). The patient was HIV-negative. Chest radiographs were normal except for a small pleural effusion. Radiographs showed a destructive lesion of T11 with an extensive paravertebral and retropleural abscess tracking a spinal level above and below with extension into the spinal canal (Figure 1).

As the patient had signs of spinal cord compression, he was taken to surgery for incision and drainage and culture procurement and corpectomy of T11 with autogenous rib graft. One week later, he was stabilized with posterior fusion and instrumentation (Figure 2). Gram stain of the specimen demonstrated broad-based budding yeast forms 15 to 20 micrometers in size, consistent with blastomycosis. Cultures were positive for Blastomyces dermatitidis. Histopathologic slides (Figure 3) of the surgical pathology specimen showed granulomatous inflammation. Oral itraconazole 200 mg twice daily was continued, as it has been found to be efficacious in treating immunocompetent patients with blastomycosis¹⁷ and is considered the medication of choice for non–life-threatening, non-CNS blastomycosis. (Intravenous amphotericin B was ruled out because of its known serious side effects, such as bone marrow suppression and renal function impairment¹⁰; itraconazole was the better alternative.) The patient was placed in a thoracolumbar orthosis and discharged. As the effect of presence of instrumentation in the setting of a fungal infection is unknown, it was deemed prudent to maintain the patient on chronic antifungal suppression. One year after surgery, computed tomography (CT) showed solid osseous bridging through the cage crossing the T11 vertebral body, from the inferior endplate of T10 through the superior endplate of T12 (Figure 4). In addition, there had been no recurrence of the spinal infection, and the patient was neurologically intact and doing well.

Discussion

North American blastomycosis (B dermatitidis) is a ubiquitous dimorphic fungus that occurs worldwide and on occasion causes serious infections in humans.^9,23,26,29 It was first characterized in 1894 by Gilchrist and Stokes (Gilchrist disease) when they recovered the fungus from the lung tissue of a patient.³ In North America, blastomycosis infections occur from central Canada to the Gulf Coast to east of the Mississippi River.^{2,5,7,8,13,14,17,21,22,24,27,29} Additional cases of the disease have been reported in Africa,^9,16,23,28 Asia,^12,19 and South America^7,8 (Table [on pages E270-E271]). Recent epidemiologic studies have linked transmission of the disease to bodies of water and have questioned previous reports of male predominance and racial preference for African Americans (Table).

Blastomycosis is acquired when inhaled fungus (airborne conidia spores) causes a primary pulmonary infection or, rarely, when there is direct inoculation through the skin. The differential diagnosis includes neoplasm, tuberculosis, actinomycosis, bacterial infections, cryptococcosis, and coccidioidomycosis.^{3,9,12,20,25,31} Blastomycosis occurs in adults and children.^1-30 The rate of mortality is much higher in immunocompromised patients. Initial symptoms include fever, chills, fatigue, malaise, myalgia, arthalgia, weight loss, and stigmata of chronic disease.^1-30 Acute pulmonary infection with blastomycosis generally resolves spontaneously but may progress to acute respiratory distress syndrome, which has a mortality rate of 50% to 89%.¹⁹ With systemic dissemination, the infection may spread to other organs¹¹—there is a particular predilection for the skin^9,20,29—and to the long bones^7,16 and the oropharynx.^16,26,28

In 50% to 64% of cases, bone involvement may be the first disease manifestation.^6,7,16,22 Osseous involvement with blastomycosis most commonly affects the long bones¹⁵ but may include the vertebrae,^1-29 the ribs,²⁶ and the carpal or tarsal bones.^7,16 The most common vertebral involvement occurs in the thoracic or lumbar spine^{1,2,7-9,11-14,17,19,21-24,26} and typically results in destruction of the body, development of a paraspinal abscess, and potential extension into the spinal canal, causing an epidural abscess and development of chronic draining cutaneous sinuses.^{2,7,9,11-13,16,17,19,22,23,26,28,29} In the present case, we do not know whether the vertebral body was involved before the patient presented with mid-thoracolumbar back pain. There may have been bony involvement during initial presentation.

Diagnosis is often difficult because of a low index of suspicion, leading to a significant delay in treatment. Primary pulmonary infections are successfully diagnosed 86% of the time from sputum and 92% of the time from bronchoscopy.¹⁹ Once the infection involves the spine, plain radiographs, CT, and magnetic resonance imaging (MRI) can be used to identify not only the bony involvement but also any adjacent soft-tissue extension.¹³ The radiographic findings, typical of tuberculosis or a neoplasm, include disc space narrowing, vertebral body destruction and collapse, late segmental kyphotic deformity, and development of a psoas abscess or a retropleural abscess.^7,26 Such abscesses lend themselves well to fine-needle aspiration,^{7,8,11,13,14,17,19,26} which, when combined with CT and MRI guidance, reliably assists in the diagnosis of blastomycosis.^1,13,17 If fine-needle aspiration fails, then open biopsy and surgical débridement specimens may be effective in the diagnosis.^{2,9,12,21,22,27}

The mortality rate for systemic blastomycosis exceeded 90% before the development of antifungal medications, and these medications remain the primary treatment for most initial infections.¹⁵ For severe infections in critically ill patients and for patients with CNS involvement, amphotericin B has been effective, with cure rates approaching 97%.¹⁷ Itraconazole, which is well tolerated, has replaced ketoconazole as the preferred long-term oral treatment for blastomycosis. Cure rates for itraconazole approach 90% when treatment is instituted over 2 years in a compliant patient.^10,19,20 Nonsurgical (antifungal) treatment for blastomycosis of the spine has also proved successful in neurologically intact patients.^7,9,11,26,28

A case involving the spine and requiring surgical drainage was first reported in 1908⁵; since then, only a few more cases have been reported.^{1,2,5,7-9,11-14,16,17,19,21-24,26-29} Thus, the literature includes very little information that can be used to establish indications for surgery for a blastomycotic infection of the spine. However, there is enough evidence to establish that surgery is indicated for patients who have a known blastomycosis infection and are developing neurologic or structural loss of integrity of the spinal column or have an abscess that requires drainage and débridement.

Our patient had been on long-term antifungal treatment but nevertheless developed a destructive spinal lesion with a concurrent epidural and retropleural abscess. Given his risk of pathologic fracture, we performed anterior débridement and stabilization followed by posterior fusion and instrumentation. We are unaware of any other cases in which an anterior titanium cage was combined with rib autograft after anterior débridement and vertebrectomy combined with posterior instrumentation for blastomycosis. This technique proved very useful, as it allowed for immediate stabilization of the spine. Therefore, the treatment goal is similar to that for any destructive infection that fails medical treatment: preservation of neurologic function, stabilization of spinal vertebrae, débridement of abscess cavity, and definitive culture procurement.

Conclusion

Although there is little reported information regarding surgical indications for blastomycotic vertebral osteomyelitis that has failed medical management—in patients with a destructive lesion and compromise of both the spinal canal and the integrity of the vertebral column—anterior débridement and stabilization followed by posterior fusion and instrumentation are useful in preventing vertebral collapse, further canal compromise, and possible cord injury.

Blastomycosis is a rare fungal infection that primarily produces acute lung infections but may on occasion disseminate to multiple sites, including the skin, bone, central nervous system (CNS), and oropharynx.^1-30 In the case of a primary infection of the lung, if there is a high index of suspicion and a thorough diagnostic workup, the diagnosis can be made from sputum or bronchoscopy.²⁴ Patients present with acute pneumonia that either resolves spontaneously or proceeds to chronic pneumonia with extrapulmonary spread to multiple organs, including the spine. Once vertebral involvement occurs, an untreated infection may result in vertebral body destruction and paraspinal and epidural abscess formation followed by neurologic injury and loss of structural integrity of the spine.^{11,13,17,23,27,29}

In this article, we present a case of blastomycosis of the vertebral body and provide a detailed review of the literature concerning this extremely rare infection of the spine. The patient provided written informed consent for print and electronic publication of this case report.

Case Report

A 30-year-old African American man with known pulmonary blastomycosis, for which he had been treated with oral itraconazole 200 mg twice daily for 6 months, was admitted to the hospital with a 2-month history of mild thoracolumbar back pain. He reported transient numbness and tingling in the lower extremities but no weakness. He denied weight loss, fatigue, appetite loss, and significant night pain. On physical examination, he was alert and oriented, well nourished, and in no acute distress. Percussion revealed limited range of motion and pain. Further examination of the spine demonstrated no spasm, swelling, erythema, or drainage. The lower extremities had intact sensation, motor strength, reflexes, and pulses, and clonus was absent. White blood cell count was 8100 cells/μL (normal), erythrocyte sedimentation rate was 77 mm/h (normal range, 0-20 mm/h), and C-reactive protein level was 57.2 mg/L (normal, ≤ 10 mg/L). The patient was HIV-negative. Chest radiographs were normal except for a small pleural effusion. Radiographs showed a destructive lesion of T11 with an extensive paravertebral and retropleural abscess tracking a spinal level above and below with extension into the spinal canal (Figure 1).

As the patient had signs of spinal cord compression, he was taken to surgery for incision and drainage and culture procurement and corpectomy of T11 with autogenous rib graft. One week later, he was stabilized with posterior fusion and instrumentation (Figure 2). Gram stain of the specimen demonstrated broad-based budding yeast forms 15 to 20 micrometers in size, consistent with blastomycosis. Cultures were positive for Blastomyces dermatitidis. Histopathologic slides (Figure 3) of the surgical pathology specimen showed granulomatous inflammation. Oral itraconazole 200 mg twice daily was continued, as it has been found to be efficacious in treating immunocompetent patients with blastomycosis¹⁷ and is considered the medication of choice for non–life-threatening, non-CNS blastomycosis. (Intravenous amphotericin B was ruled out because of its known serious side effects, such as bone marrow suppression and renal function impairment¹⁰; itraconazole was the better alternative.) The patient was placed in a thoracolumbar orthosis and discharged. As the effect of presence of instrumentation in the setting of a fungal infection is unknown, it was deemed prudent to maintain the patient on chronic antifungal suppression. One year after surgery, computed tomography (CT) showed solid osseous bridging through the cage crossing the T11 vertebral body, from the inferior endplate of T10 through the superior endplate of T12 (Figure 4). In addition, there had been no recurrence of the spinal infection, and the patient was neurologically intact and doing well.

Discussion

North American blastomycosis (B dermatitidis) is a ubiquitous dimorphic fungus that occurs worldwide and on occasion causes serious infections in humans.^9,23,26,29 It was first characterized in 1894 by Gilchrist and Stokes (Gilchrist disease) when they recovered the fungus from the lung tissue of a patient.³ In North America, blastomycosis infections occur from central Canada to the Gulf Coast to east of the Mississippi River.^{2,5,7,8,13,14,17,21,22,24,27,29} Additional cases of the disease have been reported in Africa,^9,16,23,28 Asia,^12,19 and South America^7,8 (Table [on pages E270-E271]). Recent epidemiologic studies have linked transmission of the disease to bodies of water and have questioned previous reports of male predominance and racial preference for African Americans (Table).

Blastomycosis is acquired when inhaled fungus (airborne conidia spores) causes a primary pulmonary infection or, rarely, when there is direct inoculation through the skin. The differential diagnosis includes neoplasm, tuberculosis, actinomycosis, bacterial infections, cryptococcosis, and coccidioidomycosis.^{3,9,12,20,25,31} Blastomycosis occurs in adults and children.^1-30 The rate of mortality is much higher in immunocompromised patients. Initial symptoms include fever, chills, fatigue, malaise, myalgia, arthalgia, weight loss, and stigmata of chronic disease.^1-30 Acute pulmonary infection with blastomycosis generally resolves spontaneously but may progress to acute respiratory distress syndrome, which has a mortality rate of 50% to 89%.¹⁹ With systemic dissemination, the infection may spread to other organs¹¹—there is a particular predilection for the skin^9,20,29—and to the long bones^7,16 and the oropharynx.^16,26,28

In 50% to 64% of cases, bone involvement may be the first disease manifestation.^6,7,16,22 Osseous involvement with blastomycosis most commonly affects the long bones¹⁵ but may include the vertebrae,^1-29 the ribs,²⁶ and the carpal or tarsal bones.^7,16 The most common vertebral involvement occurs in the thoracic or lumbar spine^{1,2,7-9,11-14,17,19,21-24,26} and typically results in destruction of the body, development of a paraspinal abscess, and potential extension into the spinal canal, causing an epidural abscess and development of chronic draining cutaneous sinuses.^{2,7,9,11-13,16,17,19,22,23,26,28,29} In the present case, we do not know whether the vertebral body was involved before the patient presented with mid-thoracolumbar back pain. There may have been bony involvement during initial presentation.

Diagnosis is often difficult because of a low index of suspicion, leading to a significant delay in treatment. Primary pulmonary infections are successfully diagnosed 86% of the time from sputum and 92% of the time from bronchoscopy.¹⁹ Once the infection involves the spine, plain radiographs, CT, and magnetic resonance imaging (MRI) can be used to identify not only the bony involvement but also any adjacent soft-tissue extension.¹³ The radiographic findings, typical of tuberculosis or a neoplasm, include disc space narrowing, vertebral body destruction and collapse, late segmental kyphotic deformity, and development of a psoas abscess or a retropleural abscess.^7,26 Such abscesses lend themselves well to fine-needle aspiration,^{7,8,11,13,14,17,19,26} which, when combined with CT and MRI guidance, reliably assists in the diagnosis of blastomycosis.^1,13,17 If fine-needle aspiration fails, then open biopsy and surgical débridement specimens may be effective in the diagnosis.^{2,9,12,21,22,27}

The mortality rate for systemic blastomycosis exceeded 90% before the development of antifungal medications, and these medications remain the primary treatment for most initial infections.¹⁵ For severe infections in critically ill patients and for patients with CNS involvement, amphotericin B has been effective, with cure rates approaching 97%.¹⁷ Itraconazole, which is well tolerated, has replaced ketoconazole as the preferred long-term oral treatment for blastomycosis. Cure rates for itraconazole approach 90% when treatment is instituted over 2 years in a compliant patient.^10,19,20 Nonsurgical (antifungal) treatment for blastomycosis of the spine has also proved successful in neurologically intact patients.^7,9,11,26,28

A case involving the spine and requiring surgical drainage was first reported in 1908⁵; since then, only a few more cases have been reported.^{1,2,5,7-9,11-14,16,17,19,21-24,26-29} Thus, the literature includes very little information that can be used to establish indications for surgery for a blastomycotic infection of the spine. However, there is enough evidence to establish that surgery is indicated for patients who have a known blastomycosis infection and are developing neurologic or structural loss of integrity of the spinal column or have an abscess that requires drainage and débridement.

Our patient had been on long-term antifungal treatment but nevertheless developed a destructive spinal lesion with a concurrent epidural and retropleural abscess. Given his risk of pathologic fracture, we performed anterior débridement and stabilization followed by posterior fusion and instrumentation. We are unaware of any other cases in which an anterior titanium cage was combined with rib autograft after anterior débridement and vertebrectomy combined with posterior instrumentation for blastomycosis. This technique proved very useful, as it allowed for immediate stabilization of the spine. Therefore, the treatment goal is similar to that for any destructive infection that fails medical treatment: preservation of neurologic function, stabilization of spinal vertebrae, débridement of abscess cavity, and definitive culture procurement.

Conclusion

Although there is little reported information regarding surgical indications for blastomycotic vertebral osteomyelitis that has failed medical management—in patients with a destructive lesion and compromise of both the spinal canal and the integrity of the vertebral column—anterior débridement and stabilization followed by posterior fusion and instrumentation are useful in preventing vertebral collapse, further canal compromise, and possible cord injury.

Blastomycosis is a rare fungal infection that primarily produces acute lung infections but may on occasion disseminate to multiple sites, including the skin, bone, central nervous system (CNS), and oropharynx.^1-30 In the case of a primary infection of the lung, if there is a high index of suspicion and a thorough diagnostic workup, the diagnosis can be made from sputum or bronchoscopy.²⁴ Patients present with acute pneumonia that either resolves spontaneously or proceeds to chronic pneumonia with extrapulmonary spread to multiple organs, including the spine. Once vertebral involvement occurs, an untreated infection may result in vertebral body destruction and paraspinal and epidural abscess formation followed by neurologic injury and loss of structural integrity of the spine.^{11,13,17,23,27,29}

In this article, we present a case of blastomycosis of the vertebral body and provide a detailed review of the literature concerning this extremely rare infection of the spine. The patient provided written informed consent for print and electronic publication of this case report.

Case Report

A 30-year-old African American man with known pulmonary blastomycosis, for which he had been treated with oral itraconazole 200 mg twice daily for 6 months, was admitted to the hospital with a 2-month history of mild thoracolumbar back pain. He reported transient numbness and tingling in the lower extremities but no weakness. He denied weight loss, fatigue, appetite loss, and significant night pain. On physical examination, he was alert and oriented, well nourished, and in no acute distress. Percussion revealed limited range of motion and pain. Further examination of the spine demonstrated no spasm, swelling, erythema, or drainage. The lower extremities had intact sensation, motor strength, reflexes, and pulses, and clonus was absent. White blood cell count was 8100 cells/μL (normal), erythrocyte sedimentation rate was 77 mm/h (normal range, 0-20 mm/h), and C-reactive protein level was 57.2 mg/L (normal, ≤ 10 mg/L). The patient was HIV-negative. Chest radiographs were normal except for a small pleural effusion. Radiographs showed a destructive lesion of T11 with an extensive paravertebral and retropleural abscess tracking a spinal level above and below with extension into the spinal canal (Figure 1).

As the patient had signs of spinal cord compression, he was taken to surgery for incision and drainage and culture procurement and corpectomy of T11 with autogenous rib graft. One week later, he was stabilized with posterior fusion and instrumentation (Figure 2). Gram stain of the specimen demonstrated broad-based budding yeast forms 15 to 20 micrometers in size, consistent with blastomycosis. Cultures were positive for Blastomyces dermatitidis. Histopathologic slides (Figure 3) of the surgical pathology specimen showed granulomatous inflammation. Oral itraconazole 200 mg twice daily was continued, as it has been found to be efficacious in treating immunocompetent patients with blastomycosis¹⁷ and is considered the medication of choice for non–life-threatening, non-CNS blastomycosis. (Intravenous amphotericin B was ruled out because of its known serious side effects, such as bone marrow suppression and renal function impairment¹⁰; itraconazole was the better alternative.) The patient was placed in a thoracolumbar orthosis and discharged. As the effect of presence of instrumentation in the setting of a fungal infection is unknown, it was deemed prudent to maintain the patient on chronic antifungal suppression. One year after surgery, computed tomography (CT) showed solid osseous bridging through the cage crossing the T11 vertebral body, from the inferior endplate of T10 through the superior endplate of T12 (Figure 4). In addition, there had been no recurrence of the spinal infection, and the patient was neurologically intact and doing well.

Discussion

North American blastomycosis (B dermatitidis) is a ubiquitous dimorphic fungus that occurs worldwide and on occasion causes serious infections in humans.^9,23,26,29 It was first characterized in 1894 by Gilchrist and Stokes (Gilchrist disease) when they recovered the fungus from the lung tissue of a patient.³ In North America, blastomycosis infections occur from central Canada to the Gulf Coast to east of the Mississippi River.^{2,5,7,8,13,14,17,21,22,24,27,29} Additional cases of the disease have been reported in Africa,^9,16,23,28 Asia,^12,19 and South America^7,8 (Table [on pages E270-E271]). Recent epidemiologic studies have linked transmission of the disease to bodies of water and have questioned previous reports of male predominance and racial preference for African Americans (Table).

Blastomycosis is acquired when inhaled fungus (airborne conidia spores) causes a primary pulmonary infection or, rarely, when there is direct inoculation through the skin. The differential diagnosis includes neoplasm, tuberculosis, actinomycosis, bacterial infections, cryptococcosis, and coccidioidomycosis.^{3,9,12,20,25,31} Blastomycosis occurs in adults and children.^1-30 The rate of mortality is much higher in immunocompromised patients. Initial symptoms include fever, chills, fatigue, malaise, myalgia, arthalgia, weight loss, and stigmata of chronic disease.^1-30 Acute pulmonary infection with blastomycosis generally resolves spontaneously but may progress to acute respiratory distress syndrome, which has a mortality rate of 50% to 89%.¹⁹ With systemic dissemination, the infection may spread to other organs¹¹—there is a particular predilection for the skin^9,20,29—and to the long bones^7,16 and the oropharynx.^16,26,28

In 50% to 64% of cases, bone involvement may be the first disease manifestation.^6,7,16,22 Osseous involvement with blastomycosis most commonly affects the long bones¹⁵ but may include the vertebrae,^1-29 the ribs,²⁶ and the carpal or tarsal bones.^7,16 The most common vertebral involvement occurs in the thoracic or lumbar spine^{1,2,7-9,11-14,17,19,21-24,26} and typically results in destruction of the body, development of a paraspinal abscess, and potential extension into the spinal canal, causing an epidural abscess and development of chronic draining cutaneous sinuses.^{2,7,9,11-13,16,17,19,22,23,26,28,29} In the present case, we do not know whether the vertebral body was involved before the patient presented with mid-thoracolumbar back pain. There may have been bony involvement during initial presentation.

Diagnosis is often difficult because of a low index of suspicion, leading to a significant delay in treatment. Primary pulmonary infections are successfully diagnosed 86% of the time from sputum and 92% of the time from bronchoscopy.¹⁹ Once the infection involves the spine, plain radiographs, CT, and magnetic resonance imaging (MRI) can be used to identify not only the bony involvement but also any adjacent soft-tissue extension.¹³ The radiographic findings, typical of tuberculosis or a neoplasm, include disc space narrowing, vertebral body destruction and collapse, late segmental kyphotic deformity, and development of a psoas abscess or a retropleural abscess.^7,26 Such abscesses lend themselves well to fine-needle aspiration,^{7,8,11,13,14,17,19,26} which, when combined with CT and MRI guidance, reliably assists in the diagnosis of blastomycosis.^1,13,17 If fine-needle aspiration fails, then open biopsy and surgical débridement specimens may be effective in the diagnosis.^{2,9,12,21,22,27}

The mortality rate for systemic blastomycosis exceeded 90% before the development of antifungal medications, and these medications remain the primary treatment for most initial infections.¹⁵ For severe infections in critically ill patients and for patients with CNS involvement, amphotericin B has been effective, with cure rates approaching 97%.¹⁷ Itraconazole, which is well tolerated, has replaced ketoconazole as the preferred long-term oral treatment for blastomycosis. Cure rates for itraconazole approach 90% when treatment is instituted over 2 years in a compliant patient.^10,19,20 Nonsurgical (antifungal) treatment for blastomycosis of the spine has also proved successful in neurologically intact patients.^7,9,11,26,28

A case involving the spine and requiring surgical drainage was first reported in 1908⁵; since then, only a few more cases have been reported.^{1,2,5,7-9,11-14,16,17,19,21-24,26-29} Thus, the literature includes very little information that can be used to establish indications for surgery for a blastomycotic infection of the spine. However, there is enough evidence to establish that surgery is indicated for patients who have a known blastomycosis infection and are developing neurologic or structural loss of integrity of the spinal column or have an abscess that requires drainage and débridement.

Our patient had been on long-term antifungal treatment but nevertheless developed a destructive spinal lesion with a concurrent epidural and retropleural abscess. Given his risk of pathologic fracture, we performed anterior débridement and stabilization followed by posterior fusion and instrumentation. We are unaware of any other cases in which an anterior titanium cage was combined with rib autograft after anterior débridement and vertebrectomy combined with posterior instrumentation for blastomycosis. This technique proved very useful, as it allowed for immediate stabilization of the spine. Therefore, the treatment goal is similar to that for any destructive infection that fails medical treatment: preservation of neurologic function, stabilization of spinal vertebrae, débridement of abscess cavity, and definitive culture procurement.

Conclusion

Although there is little reported information regarding surgical indications for blastomycotic vertebral osteomyelitis that has failed medical management—in patients with a destructive lesion and compromise of both the spinal canal and the integrity of the vertebral column—anterior débridement and stabilization followed by posterior fusion and instrumentation are useful in preventing vertebral collapse, further canal compromise, and possible cord injury.

Assessment of cervical spine range of motion (ROM) is an integral aspect of the physical examination for cervical conditions,^1-3 surgical outcomes,⁴ and functional impairment.¹ In fact, the emphasis being placed on such functional measures before and after treatments is increasing.^4,5

Cervical spine range of motion is routinely used as an outcome measure in clinical studies.^6-8 Underscoring the importance of defining cervical spine ROM, studies have found it to be a preoperative predictor of outcomes of anterior cervical surgery,⁹ and other studies have suggested it is a determinant of athletes’ return to play.¹⁰

Spinal ROM measurements can be used to determine the degree of disability experienced by a patient with a spinal condition as defined in the Guides to the Evaluation of Permanent Impairment by the American Medical Association (AMA).¹ In the medicolegal realm, ROM measurements made by clinicians can influence the dollar amounts of awards in legal claims, and, according to the AMA guides, the difference in cervical spine ROM between normality and disability or impairment can be as little as 5°.

Although cervical spine ROM is routinely assessed and documented in clinical practice, no universal protocol exists for its evaluation.^11,12 In fact, considerable inter-examiner variation in visual estimates of ROM has been found,^13-16 and significant inaccuracies have been reported.^17,18

Goniometers have been shown to be reliable and highly accurate, with low inter-examiner and intra-examiner variability.^5,19-21 Nevertheless, logistics²² and costs²¹ generally limit their being accepted in routine clinical practice. Among many methods available for assessing ROM, visual estimation is the least reliable or accurate,²³ but it is the quickest and least expensive and is recommended in textbooks that describe the spinal-specific physical examination.²⁴ Despite the superiority of goniometers in measuring ROM, these significant barriers have limited their use in clinical practice. When assessing cervical spine ROM, most clinicians prefer visual estimates over goniometers.

We conducted a study to determine whether training could improve the accuracy of visual estimates. We compared the accuracy of visual estimates of cervical spine ROM with that of a radiographically validated electrogoniometer and then investigated whether accuracy and reliability of visual estimates could be improved with a session of instruction and demonstration. Assessments of accuracy were made immediately after and 1 month after this training session.

Materials and Methods

Assessments Made Before Training

This study was approved by our institution’s human investigation committee and was conducted in accordance with the ethical standards of that committee.

Cervical spine ROM was assessed by 8 examiners (2 attending spine surgeons, 4 orthopedic residents, 2 medical students). They were informed they would be participating in a study evaluating visual estimates of motion but were given no other information prior to the study.

Four healthy volunteer subjects (examiners who rotated through the role) were assessed. No subject reported any ongoing neck or spine discomfort or had had any previous spinal surgery. One at a time, subjects were fitted with a cervical harness electrogoniometer capable of measuring angulation of the cervical spine to the nearest degree (modified electrogoniometer, torsiometer, and display from Biometrics, Gwent, UK; Figures 1A, 1B). This electrogoniometer has been shown to have a mean (SD) error of 2.3° (2.6°) relative to radiographic assessments.⁸

With the electrogoniometer fitted, each subject was instructed to sit upright in a chair with his back to the backrest and his head neutrally positioned. The electrogoniometer was then zeroed, and the subject proceeded with 5 series of flexion-extension, left and right lateral bending, and left and right rotation movements. The subject was instructed to make 1 movement in full motion in each direction and the other 4 movements in less than full motion to yield a variety of excursions for assessment. Each subject was instructed to pause at the apex of each motion. During these pauses, the examiners recorded their visual estimates of movement in each direction while the investigator recorded degrees of motion (displayed by the electrogoniometer) in flexion-extension, lateral bending, and rotation (Figures 2A–2D). The electrogoniometer display was not visible to subjects or examiners.

A total of 840 independent visual estimates of 120 distinct movements were recorded.

Training, and Assessments Made Immediately Thereafter

After the first round of visual estimates, the 8 examiners were verbally instructed in cervical spine ROM assessment and were asked to observe 1 subject, fitted with the electrogoniometer, demonstrating partial and full cervical motions while the investigator announced the electrogoniometric measurements. The motions demonstrated included 15°, 30°, and the extremes of cervical spine ROM in each of 6 directions from neutral.

After this training session, each of the 4 subjects from the first round of assessments was again fitted with the harness electrogoniometer and instructed to repeat the movements in turn while examiners visually estimated cervical spine ROM and independently recorded their estimates. Meanwhile, the investigator recorded the degree of motion during each movement (as measured by the electrogoniometer). Again, a total of 840 independent visual estimates of 120 distinct movements were recorded.

Assessments Made 1 Month After Training

One month after the training session, the examiners and the investigator reconvened to assess the same 4 subjects using a procedure for simultaneous visual estimation and electrogoniometric measurement identical to that used 1 month earlier. No additional training was given. Again, 840 independent visual estimates of 120 distinct movements were recorded.

Data Analysis

The reliabilities of visual estimates were analyzed by calculating the intraclass coefficients (ICCs) using random-effect 1-way analyses of variance. By convention, ICCs of < 0.2, 0.2 to 0.39, 0.4 to 0.59, 0.6 to 0.8, and > 0.8 correspond to poor, fair, moderate, substantial, and perfect reliability, respectively.²⁵

We compared the visual estimates and electrogoniometric measurements made for 3 planes of motion (flexion-extension, lateral bending, axial rotation) before, immediately after, and 1 month after training and drew trend lines generated by linear regression relative to a line of perfect correlation.

Mean errors in examiners’ visual estimates (relative to elec­trogoniometric measurements) made before, immediately after, and 1 month after training were calculated. Paired Student t tests were then used to compare the mean errors before training with the mean errors immediately after and 1 month after training.

All analyses were performed with SPSS for Windows 16.0 (SPSS, Chicago, Illinois).

Results

Inter-examiner reliability of the visual estimates in all planes of motion ranged from 0.51 to 0.79 (suggestive of moderate to substantial reliability). For reference, standard goniometers measuring knee ROM have inter-examiner ICCs of 0.89 to 0.98²⁶ (suggestive of perfect reliability). The ICCs before, immediately after, and 1 month after training were not significantly different. 

As expected, there were significant errors in visual estimates of cervical spine ROM in all planes. Initial errors in visual estimates (relative to electrogoniometric measurements) were 23.9° (flexion-extension), 15.5° (lateral bending), and 19.3° (axial rotation) (Table, Figure 3).

Immediately after training, mean errors in visual estimates decreased to 12.0° (flexion-extension), 11.7° (lateral bending), and 16.4° (axial rotation) (Table, Figure 3). In all 3 planes of cervical motion, these improvements were statistically significant.

One month after training, mean errors in visual estimates were 14.4° (flexion-extension), 13.9° (lateral bending), and 16.2° (axial rotation) (Table, Figure 3). Only the improvement in the estimate of flexion-extension (the direction of the largest error initially) remained statistically significant—a 39.7% decrease in error.

We also considered how errors varied with degree of motion observed. In flexion-extension, the tendency to overestimate at larger degrees of motion was not apparent after training, and 1 month after training we found a tendency to underestimate at smaller degrees of motion (Figure 4A). The tendency to overestimate lateral bending before training did not persist immediately after or 1 month after training (Figure 4B). Estimates of axial rotation correlated well with goniometer measurements before training and were also well correlated immediately after and 1 month after training (Figure 4C).

Discussion

Visual estimation of spinal motion is unreliable and inaccurate, but its widespread use in clinical practice continues. Goniometers are far more accurate and reliable but are seldom used. We investigated whether a training session featuring verbal instruction and demonstration with an electrogoniometer could improve visual estimates and whether potential improvement in visual estimates would remain 1 month after training.

Widely variable ICCs (0.42-0.90) have been reported for visual estimates of cervical spine ROM.^17,18,22 Our findings on the reliability of these estimates are consistent with the literature.

We recorded the greatest initial error in estimates of motion in flexion-extension. Previous studies have also found the greatest error and least reliability in visual estimates in this plane.^14,15,18 Visual estimation may be more difficult in flexion-extension because the shoulders cannot be used as landmarks, whereas they serve as approximate 90° reference points during estimation of lateral bending and axial rotation. Demonstration of 15°, 30° and the extremes of ROM during the training session may have provided alternative reference points during visual estimation after training—decreasing the error to within the range found in other planes of motion.

Initial errors in visual estimates were 23.9° (flexion-extension), 15.5° (lateral bending), and 19.3° (axial rotation). Based on normative cervical spine ROM in a healthy population— 126° ± 12° for flexion-extension, 86° ± 5° for lateral bending, 151° ± 23° for axial rotation²²—the errors we identified are 18.9% of the normal range of flexion-extension, 18.0% of lateral bending, and 12.8% of axial rotation.

Training clearly improved the accuracy of visual estimates of cervical spine ROM. Estimates were statistically improved for all planes immediately after training and remained significantly improved for flexion-extension (the plane of largest error initially) 1 month after training. Before training, mean errors varied across planes. Training normalized mean errors to about 15°, and this effect lasted in flexion-extension, lateral bending, and axial rotation (Figures 4A–4C). Of note, before training these percentage errors increased with increased motion from neutral in the flexion-extension and lateral bending planes. At full ROM, percentage errors in estimates were greater. After training, percentage errors did not increase appreciably with increasing motion.

Readers will naturally reflect on the clinical significance of the motion assessment improvements demonstrated after the training session described in this study. We must be aware that functional assessments are increasingly being emphasized in the clinical arena—with respect to clinical conditions, surgical outcomes, and functional impairments. We highlight a point made earlier: A difference of only 5° can affect impairment ratings in the medicolegal realm.¹ In estimating flexion-extension motion, lasting improvements of almost 10° were demonstrated and maintained 1 month after the training session described in this study.

Nevertheless, mean errors in visual estimation remained at about 15° in all planes of motion, despite our modest improvements. This finding raises the question of whether visually estimated ROM should be pertinent to assessments of impairment and disability. Although visual estimates of ROM may have more utility as a screening test for impairment and disability, fine differences in ROM simply cannot be reliably assessed by visual estimation.

This study has limitations. First, it was conducted at a single institution where the evaluators received most of their training. Their skill in visually estimating cervical spine ROM may not be generalizable to a larger population of spine specialists who are practicing at other institutions and may have different training backgrounds.

Second, only healthy subjects were assessed. Some studies of cervical spine ROM have shown better reliability in symptomatic subjects relative to asymptomatic subjects.^13,14 To attempt to overcome this limitation, we assessed many different excursions of motion that were often not to the extremes of motion.

Third, the “gold standard” we used for motion assessment was an electrogoniometer, which has some inherent error (previously validated mean [SD] error of 2.3° [2.6°] relative to radiographs⁸). Although obtaining radiographs of each movement would have more closely resembled the gold standard, the radiation dose associated with such a study is prohibitive.

Last, the assessors included medical students. The medical students’ estimates, however, tended to be more accurate than the residents’ or attending surgeons’ (though the difference was not statistically significant). This tendency may reflect the medical students’ closer attention to detail.  Clearly, including medical students in the study did not negatively affect the accuracy of the estimates or the validity of our findings.

Conclusion

Despite its limitations, visual assessment of cervical spine motion remains the gold standard in clinical practice and is routinely recorded and reported. Mean errors ranged from 15.5° to 23.9°, depending on plane of motion being assessed, but these improved after a training session.

Visual estimates of motion in flexion-extension were most improved by training, as the initial errors in this plane were the largest. Statistically significant improvement of about 10° remained for flexion-extension motion estimates 1 month after training.

During a time when we are increasingly emphasizing functional outcomes, such a degree of improvement could be of clinical significance. Our study results support a call for more formalized training of ROM assessment, but clinicians should also be aware of the limitations of visual estimates of cervical spine ROM, and our study results support scrutiny of visual assessment of ROM as a criterion for diagnosing permanent impairment or disability.

Assessment of cervical spine range of motion (ROM) is an integral aspect of the physical examination for cervical conditions,^1-3 surgical outcomes,⁴ and functional impairment.¹ In fact, the emphasis being placed on such functional measures before and after treatments is increasing.^4,5

Cervical spine range of motion is routinely used as an outcome measure in clinical studies.^6-8 Underscoring the importance of defining cervical spine ROM, studies have found it to be a preoperative predictor of outcomes of anterior cervical surgery,⁹ and other studies have suggested it is a determinant of athletes’ return to play.¹⁰

Spinal ROM measurements can be used to determine the degree of disability experienced by a patient with a spinal condition as defined in the Guides to the Evaluation of Permanent Impairment by the American Medical Association (AMA).¹ In the medicolegal realm, ROM measurements made by clinicians can influence the dollar amounts of awards in legal claims, and, according to the AMA guides, the difference in cervical spine ROM between normality and disability or impairment can be as little as 5°.

Although cervical spine ROM is routinely assessed and documented in clinical practice, no universal protocol exists for its evaluation.^11,12 In fact, considerable inter-examiner variation in visual estimates of ROM has been found,^13-16 and significant inaccuracies have been reported.^17,18

Goniometers have been shown to be reliable and highly accurate, with low inter-examiner and intra-examiner variability.^5,19-21 Nevertheless, logistics²² and costs²¹ generally limit their being accepted in routine clinical practice. Among many methods available for assessing ROM, visual estimation is the least reliable or accurate,²³ but it is the quickest and least expensive and is recommended in textbooks that describe the spinal-specific physical examination.²⁴ Despite the superiority of goniometers in measuring ROM, these significant barriers have limited their use in clinical practice. When assessing cervical spine ROM, most clinicians prefer visual estimates over goniometers.

We conducted a study to determine whether training could improve the accuracy of visual estimates. We compared the accuracy of visual estimates of cervical spine ROM with that of a radiographically validated electrogoniometer and then investigated whether accuracy and reliability of visual estimates could be improved with a session of instruction and demonstration. Assessments of accuracy were made immediately after and 1 month after this training session.

Materials and Methods

Assessments Made Before Training

This study was approved by our institution’s human investigation committee and was conducted in accordance with the ethical standards of that committee.

Cervical spine ROM was assessed by 8 examiners (2 attending spine surgeons, 4 orthopedic residents, 2 medical students). They were informed they would be participating in a study evaluating visual estimates of motion but were given no other information prior to the study.

Four healthy volunteer subjects (examiners who rotated through the role) were assessed. No subject reported any ongoing neck or spine discomfort or had had any previous spinal surgery. One at a time, subjects were fitted with a cervical harness electrogoniometer capable of measuring angulation of the cervical spine to the nearest degree (modified electrogoniometer, torsiometer, and display from Biometrics, Gwent, UK; Figures 1A, 1B). This electrogoniometer has been shown to have a mean (SD) error of 2.3° (2.6°) relative to radiographic assessments.⁸

With the electrogoniometer fitted, each subject was instructed to sit upright in a chair with his back to the backrest and his head neutrally positioned. The electrogoniometer was then zeroed, and the subject proceeded with 5 series of flexion-extension, left and right lateral bending, and left and right rotation movements. The subject was instructed to make 1 movement in full motion in each direction and the other 4 movements in less than full motion to yield a variety of excursions for assessment. Each subject was instructed to pause at the apex of each motion. During these pauses, the examiners recorded their visual estimates of movement in each direction while the investigator recorded degrees of motion (displayed by the electrogoniometer) in flexion-extension, lateral bending, and rotation (Figures 2A–2D). The electrogoniometer display was not visible to subjects or examiners.

A total of 840 independent visual estimates of 120 distinct movements were recorded.

Training, and Assessments Made Immediately Thereafter

After the first round of visual estimates, the 8 examiners were verbally instructed in cervical spine ROM assessment and were asked to observe 1 subject, fitted with the electrogoniometer, demonstrating partial and full cervical motions while the investigator announced the electrogoniometric measurements. The motions demonstrated included 15°, 30°, and the extremes of cervical spine ROM in each of 6 directions from neutral.

After this training session, each of the 4 subjects from the first round of assessments was again fitted with the harness electrogoniometer and instructed to repeat the movements in turn while examiners visually estimated cervical spine ROM and independently recorded their estimates. Meanwhile, the investigator recorded the degree of motion during each movement (as measured by the electrogoniometer). Again, a total of 840 independent visual estimates of 120 distinct movements were recorded.

Assessments Made 1 Month After Training

One month after the training session, the examiners and the investigator reconvened to assess the same 4 subjects using a procedure for simultaneous visual estimation and electrogoniometric measurement identical to that used 1 month earlier. No additional training was given. Again, 840 independent visual estimates of 120 distinct movements were recorded.

Data Analysis

The reliabilities of visual estimates were analyzed by calculating the intraclass coefficients (ICCs) using random-effect 1-way analyses of variance. By convention, ICCs of < 0.2, 0.2 to 0.39, 0.4 to 0.59, 0.6 to 0.8, and > 0.8 correspond to poor, fair, moderate, substantial, and perfect reliability, respectively.²⁵

We compared the visual estimates and electrogoniometric measurements made for 3 planes of motion (flexion-extension, lateral bending, axial rotation) before, immediately after, and 1 month after training and drew trend lines generated by linear regression relative to a line of perfect correlation.

Mean errors in examiners’ visual estimates (relative to elec­trogoniometric measurements) made before, immediately after, and 1 month after training were calculated. Paired Student t tests were then used to compare the mean errors before training with the mean errors immediately after and 1 month after training.

All analyses were performed with SPSS for Windows 16.0 (SPSS, Chicago, Illinois).

Results

Inter-examiner reliability of the visual estimates in all planes of motion ranged from 0.51 to 0.79 (suggestive of moderate to substantial reliability). For reference, standard goniometers measuring knee ROM have inter-examiner ICCs of 0.89 to 0.98²⁶ (suggestive of perfect reliability). The ICCs before, immediately after, and 1 month after training were not significantly different. 

As expected, there were significant errors in visual estimates of cervical spine ROM in all planes. Initial errors in visual estimates (relative to electrogoniometric measurements) were 23.9° (flexion-extension), 15.5° (lateral bending), and 19.3° (axial rotation) (Table, Figure 3).

Immediately after training, mean errors in visual estimates decreased to 12.0° (flexion-extension), 11.7° (lateral bending), and 16.4° (axial rotation) (Table, Figure 3). In all 3 planes of cervical motion, these improvements were statistically significant.

One month after training, mean errors in visual estimates were 14.4° (flexion-extension), 13.9° (lateral bending), and 16.2° (axial rotation) (Table, Figure 3). Only the improvement in the estimate of flexion-extension (the direction of the largest error initially) remained statistically significant—a 39.7% decrease in error.

We also considered how errors varied with degree of motion observed. In flexion-extension, the tendency to overestimate at larger degrees of motion was not apparent after training, and 1 month after training we found a tendency to underestimate at smaller degrees of motion (Figure 4A). The tendency to overestimate lateral bending before training did not persist immediately after or 1 month after training (Figure 4B). Estimates of axial rotation correlated well with goniometer measurements before training and were also well correlated immediately after and 1 month after training (Figure 4C).

Discussion

Visual estimation of spinal motion is unreliable and inaccurate, but its widespread use in clinical practice continues. Goniometers are far more accurate and reliable but are seldom used. We investigated whether a training session featuring verbal instruction and demonstration with an electrogoniometer could improve visual estimates and whether potential improvement in visual estimates would remain 1 month after training.

Widely variable ICCs (0.42-0.90) have been reported for visual estimates of cervical spine ROM.^17,18,22 Our findings on the reliability of these estimates are consistent with the literature.

We recorded the greatest initial error in estimates of motion in flexion-extension. Previous studies have also found the greatest error and least reliability in visual estimates in this plane.^14,15,18 Visual estimation may be more difficult in flexion-extension because the shoulders cannot be used as landmarks, whereas they serve as approximate 90° reference points during estimation of lateral bending and axial rotation. Demonstration of 15°, 30° and the extremes of ROM during the training session may have provided alternative reference points during visual estimation after training—decreasing the error to within the range found in other planes of motion.

Initial errors in visual estimates were 23.9° (flexion-extension), 15.5° (lateral bending), and 19.3° (axial rotation). Based on normative cervical spine ROM in a healthy population— 126° ± 12° for flexion-extension, 86° ± 5° for lateral bending, 151° ± 23° for axial rotation²²—the errors we identified are 18.9% of the normal range of flexion-extension, 18.0% of lateral bending, and 12.8% of axial rotation.

Training clearly improved the accuracy of visual estimates of cervical spine ROM. Estimates were statistically improved for all planes immediately after training and remained significantly improved for flexion-extension (the plane of largest error initially) 1 month after training. Before training, mean errors varied across planes. Training normalized mean errors to about 15°, and this effect lasted in flexion-extension, lateral bending, and axial rotation (Figures 4A–4C). Of note, before training these percentage errors increased with increased motion from neutral in the flexion-extension and lateral bending planes. At full ROM, percentage errors in estimates were greater. After training, percentage errors did not increase appreciably with increasing motion.

Readers will naturally reflect on the clinical significance of the motion assessment improvements demonstrated after the training session described in this study. We must be aware that functional assessments are increasingly being emphasized in the clinical arena—with respect to clinical conditions, surgical outcomes, and functional impairments. We highlight a point made earlier: A difference of only 5° can affect impairment ratings in the medicolegal realm.¹ In estimating flexion-extension motion, lasting improvements of almost 10° were demonstrated and maintained 1 month after the training session described in this study.

Nevertheless, mean errors in visual estimation remained at about 15° in all planes of motion, despite our modest improvements. This finding raises the question of whether visually estimated ROM should be pertinent to assessments of impairment and disability. Although visual estimates of ROM may have more utility as a screening test for impairment and disability, fine differences in ROM simply cannot be reliably assessed by visual estimation.

This study has limitations. First, it was conducted at a single institution where the evaluators received most of their training. Their skill in visually estimating cervical spine ROM may not be generalizable to a larger population of spine specialists who are practicing at other institutions and may have different training backgrounds.

Second, only healthy subjects were assessed. Some studies of cervical spine ROM have shown better reliability in symptomatic subjects relative to asymptomatic subjects.^13,14 To attempt to overcome this limitation, we assessed many different excursions of motion that were often not to the extremes of motion.

Third, the “gold standard” we used for motion assessment was an electrogoniometer, which has some inherent error (previously validated mean [SD] error of 2.3° [2.6°] relative to radiographs⁸). Although obtaining radiographs of each movement would have more closely resembled the gold standard, the radiation dose associated with such a study is prohibitive.

Last, the assessors included medical students. The medical students’ estimates, however, tended to be more accurate than the residents’ or attending surgeons’ (though the difference was not statistically significant). This tendency may reflect the medical students’ closer attention to detail.  Clearly, including medical students in the study did not negatively affect the accuracy of the estimates or the validity of our findings.

Conclusion

Despite its limitations, visual assessment of cervical spine motion remains the gold standard in clinical practice and is routinely recorded and reported. Mean errors ranged from 15.5° to 23.9°, depending on plane of motion being assessed, but these improved after a training session.

Visual estimates of motion in flexion-extension were most improved by training, as the initial errors in this plane were the largest. Statistically significant improvement of about 10° remained for flexion-extension motion estimates 1 month after training.

During a time when we are increasingly emphasizing functional outcomes, such a degree of improvement could be of clinical significance. Our study results support a call for more formalized training of ROM assessment, but clinicians should also be aware of the limitations of visual estimates of cervical spine ROM, and our study results support scrutiny of visual assessment of ROM as a criterion for diagnosing permanent impairment or disability.

Assessment of cervical spine range of motion (ROM) is an integral aspect of the physical examination for cervical conditions,^1-3 surgical outcomes,⁴ and functional impairment.¹ In fact, the emphasis being placed on such functional measures before and after treatments is increasing.^4,5

Cervical spine range of motion is routinely used as an outcome measure in clinical studies.^6-8 Underscoring the importance of defining cervical spine ROM, studies have found it to be a preoperative predictor of outcomes of anterior cervical surgery,⁹ and other studies have suggested it is a determinant of athletes’ return to play.¹⁰

Spinal ROM measurements can be used to determine the degree of disability experienced by a patient with a spinal condition as defined in the Guides to the Evaluation of Permanent Impairment by the American Medical Association (AMA).¹ In the medicolegal realm, ROM measurements made by clinicians can influence the dollar amounts of awards in legal claims, and, according to the AMA guides, the difference in cervical spine ROM between normality and disability or impairment can be as little as 5°.

Although cervical spine ROM is routinely assessed and documented in clinical practice, no universal protocol exists for its evaluation.^11,12 In fact, considerable inter-examiner variation in visual estimates of ROM has been found,^13-16 and significant inaccuracies have been reported.^17,18

Goniometers have been shown to be reliable and highly accurate, with low inter-examiner and intra-examiner variability.^5,19-21 Nevertheless, logistics²² and costs²¹ generally limit their being accepted in routine clinical practice. Among many methods available for assessing ROM, visual estimation is the least reliable or accurate,²³ but it is the quickest and least expensive and is recommended in textbooks that describe the spinal-specific physical examination.²⁴ Despite the superiority of goniometers in measuring ROM, these significant barriers have limited their use in clinical practice. When assessing cervical spine ROM, most clinicians prefer visual estimates over goniometers.

We conducted a study to determine whether training could improve the accuracy of visual estimates. We compared the accuracy of visual estimates of cervical spine ROM with that of a radiographically validated electrogoniometer and then investigated whether accuracy and reliability of visual estimates could be improved with a session of instruction and demonstration. Assessments of accuracy were made immediately after and 1 month after this training session.

Materials and Methods

Assessments Made Before Training

This study was approved by our institution’s human investigation committee and was conducted in accordance with the ethical standards of that committee.

Cervical spine ROM was assessed by 8 examiners (2 attending spine surgeons, 4 orthopedic residents, 2 medical students). They were informed they would be participating in a study evaluating visual estimates of motion but were given no other information prior to the study.

Four healthy volunteer subjects (examiners who rotated through the role) were assessed. No subject reported any ongoing neck or spine discomfort or had had any previous spinal surgery. One at a time, subjects were fitted with a cervical harness electrogoniometer capable of measuring angulation of the cervical spine to the nearest degree (modified electrogoniometer, torsiometer, and display from Biometrics, Gwent, UK; Figures 1A, 1B). This electrogoniometer has been shown to have a mean (SD) error of 2.3° (2.6°) relative to radiographic assessments.⁸

With the electrogoniometer fitted, each subject was instructed to sit upright in a chair with his back to the backrest and his head neutrally positioned. The electrogoniometer was then zeroed, and the subject proceeded with 5 series of flexion-extension, left and right lateral bending, and left and right rotation movements. The subject was instructed to make 1 movement in full motion in each direction and the other 4 movements in less than full motion to yield a variety of excursions for assessment. Each subject was instructed to pause at the apex of each motion. During these pauses, the examiners recorded their visual estimates of movement in each direction while the investigator recorded degrees of motion (displayed by the electrogoniometer) in flexion-extension, lateral bending, and rotation (Figures 2A–2D). The electrogoniometer display was not visible to subjects or examiners.

A total of 840 independent visual estimates of 120 distinct movements were recorded.

Training, and Assessments Made Immediately Thereafter

After the first round of visual estimates, the 8 examiners were verbally instructed in cervical spine ROM assessment and were asked to observe 1 subject, fitted with the electrogoniometer, demonstrating partial and full cervical motions while the investigator announced the electrogoniometric measurements. The motions demonstrated included 15°, 30°, and the extremes of cervical spine ROM in each of 6 directions from neutral.

After this training session, each of the 4 subjects from the first round of assessments was again fitted with the harness electrogoniometer and instructed to repeat the movements in turn while examiners visually estimated cervical spine ROM and independently recorded their estimates. Meanwhile, the investigator recorded the degree of motion during each movement (as measured by the electrogoniometer). Again, a total of 840 independent visual estimates of 120 distinct movements were recorded.

Assessments Made 1 Month After Training

One month after the training session, the examiners and the investigator reconvened to assess the same 4 subjects using a procedure for simultaneous visual estimation and electrogoniometric measurement identical to that used 1 month earlier. No additional training was given. Again, 840 independent visual estimates of 120 distinct movements were recorded.

Data Analysis

The reliabilities of visual estimates were analyzed by calculating the intraclass coefficients (ICCs) using random-effect 1-way analyses of variance. By convention, ICCs of < 0.2, 0.2 to 0.39, 0.4 to 0.59, 0.6 to 0.8, and > 0.8 correspond to poor, fair, moderate, substantial, and perfect reliability, respectively.²⁵

We compared the visual estimates and electrogoniometric measurements made for 3 planes of motion (flexion-extension, lateral bending, axial rotation) before, immediately after, and 1 month after training and drew trend lines generated by linear regression relative to a line of perfect correlation.

Mean errors in examiners’ visual estimates (relative to elec­trogoniometric measurements) made before, immediately after, and 1 month after training were calculated. Paired Student t tests were then used to compare the mean errors before training with the mean errors immediately after and 1 month after training.

All analyses were performed with SPSS for Windows 16.0 (SPSS, Chicago, Illinois).

Results

Inter-examiner reliability of the visual estimates in all planes of motion ranged from 0.51 to 0.79 (suggestive of moderate to substantial reliability). For reference, standard goniometers measuring knee ROM have inter-examiner ICCs of 0.89 to 0.98²⁶ (suggestive of perfect reliability). The ICCs before, immediately after, and 1 month after training were not significantly different. 

As expected, there were significant errors in visual estimates of cervical spine ROM in all planes. Initial errors in visual estimates (relative to electrogoniometric measurements) were 23.9° (flexion-extension), 15.5° (lateral bending), and 19.3° (axial rotation) (Table, Figure 3).

Immediately after training, mean errors in visual estimates decreased to 12.0° (flexion-extension), 11.7° (lateral bending), and 16.4° (axial rotation) (Table, Figure 3). In all 3 planes of cervical motion, these improvements were statistically significant.

One month after training, mean errors in visual estimates were 14.4° (flexion-extension), 13.9° (lateral bending), and 16.2° (axial rotation) (Table, Figure 3). Only the improvement in the estimate of flexion-extension (the direction of the largest error initially) remained statistically significant—a 39.7% decrease in error.

We also considered how errors varied with degree of motion observed. In flexion-extension, the tendency to overestimate at larger degrees of motion was not apparent after training, and 1 month after training we found a tendency to underestimate at smaller degrees of motion (Figure 4A). The tendency to overestimate lateral bending before training did not persist immediately after or 1 month after training (Figure 4B). Estimates of axial rotation correlated well with goniometer measurements before training and were also well correlated immediately after and 1 month after training (Figure 4C).

Discussion

Visual estimation of spinal motion is unreliable and inaccurate, but its widespread use in clinical practice continues. Goniometers are far more accurate and reliable but are seldom used. We investigated whether a training session featuring verbal instruction and demonstration with an electrogoniometer could improve visual estimates and whether potential improvement in visual estimates would remain 1 month after training.

Widely variable ICCs (0.42-0.90) have been reported for visual estimates of cervical spine ROM.^17,18,22 Our findings on the reliability of these estimates are consistent with the literature.

We recorded the greatest initial error in estimates of motion in flexion-extension. Previous studies have also found the greatest error and least reliability in visual estimates in this plane.^14,15,18 Visual estimation may be more difficult in flexion-extension because the shoulders cannot be used as landmarks, whereas they serve as approximate 90° reference points during estimation of lateral bending and axial rotation. Demonstration of 15°, 30° and the extremes of ROM during the training session may have provided alternative reference points during visual estimation after training—decreasing the error to within the range found in other planes of motion.

Initial errors in visual estimates were 23.9° (flexion-extension), 15.5° (lateral bending), and 19.3° (axial rotation). Based on normative cervical spine ROM in a healthy population— 126° ± 12° for flexion-extension, 86° ± 5° for lateral bending, 151° ± 23° for axial rotation²²—the errors we identified are 18.9% of the normal range of flexion-extension, 18.0% of lateral bending, and 12.8% of axial rotation.

Training clearly improved the accuracy of visual estimates of cervical spine ROM. Estimates were statistically improved for all planes immediately after training and remained significantly improved for flexion-extension (the plane of largest error initially) 1 month after training. Before training, mean errors varied across planes. Training normalized mean errors to about 15°, and this effect lasted in flexion-extension, lateral bending, and axial rotation (Figures 4A–4C). Of note, before training these percentage errors increased with increased motion from neutral in the flexion-extension and lateral bending planes. At full ROM, percentage errors in estimates were greater. After training, percentage errors did not increase appreciably with increasing motion.

Readers will naturally reflect on the clinical significance of the motion assessment improvements demonstrated after the training session described in this study. We must be aware that functional assessments are increasingly being emphasized in the clinical arena—with respect to clinical conditions, surgical outcomes, and functional impairments. We highlight a point made earlier: A difference of only 5° can affect impairment ratings in the medicolegal realm.¹ In estimating flexion-extension motion, lasting improvements of almost 10° were demonstrated and maintained 1 month after the training session described in this study.

Nevertheless, mean errors in visual estimation remained at about 15° in all planes of motion, despite our modest improvements. This finding raises the question of whether visually estimated ROM should be pertinent to assessments of impairment and disability. Although visual estimates of ROM may have more utility as a screening test for impairment and disability, fine differences in ROM simply cannot be reliably assessed by visual estimation.

This study has limitations. First, it was conducted at a single institution where the evaluators received most of their training. Their skill in visually estimating cervical spine ROM may not be generalizable to a larger population of spine specialists who are practicing at other institutions and may have different training backgrounds.

Second, only healthy subjects were assessed. Some studies of cervical spine ROM have shown better reliability in symptomatic subjects relative to asymptomatic subjects.^13,14 To attempt to overcome this limitation, we assessed many different excursions of motion that were often not to the extremes of motion.

Third, the “gold standard” we used for motion assessment was an electrogoniometer, which has some inherent error (previously validated mean [SD] error of 2.3° [2.6°] relative to radiographs⁸). Although obtaining radiographs of each movement would have more closely resembled the gold standard, the radiation dose associated with such a study is prohibitive.

Last, the assessors included medical students. The medical students’ estimates, however, tended to be more accurate than the residents’ or attending surgeons’ (though the difference was not statistically significant). This tendency may reflect the medical students’ closer attention to detail.  Clearly, including medical students in the study did not negatively affect the accuracy of the estimates or the validity of our findings.

Conclusion

Despite its limitations, visual assessment of cervical spine motion remains the gold standard in clinical practice and is routinely recorded and reported. Mean errors ranged from 15.5° to 23.9°, depending on plane of motion being assessed, but these improved after a training session.

Visual estimates of motion in flexion-extension were most improved by training, as the initial errors in this plane were the largest. Statistically significant improvement of about 10° remained for flexion-extension motion estimates 1 month after training.

During a time when we are increasingly emphasizing functional outcomes, such a degree of improvement could be of clinical significance. Our study results support a call for more formalized training of ROM assessment, but clinicians should also be aware of the limitations of visual estimates of cervical spine ROM, and our study results support scrutiny of visual assessment of ROM as a criterion for diagnosing permanent impairment or disability.

Metal-on-metal hip resurfacing arthroplasty (HRA) remains an alternative to total hip arthroplasty (THA) in appropriately selected, younger, active adults with degenerative hip disease.^1-4 While concerns remain regarding the potential for adverse local tissue reactions from wear of the metal-on-metal bearing surface,^5-8 10-year data from the Australian Orthopaedic Association National Joint Replacement Registry Annual Report⁹ showed a revision rate of only 6.3% when the Birmingham Hip Resurfacing (BHR) System was used (Smith & Nephew Inc, Memphis, Tennessee).In addition, in an independent review of 230 consecutive BHRs at a mean follow-up of 10.4 years, Coulter and colleagues¹⁰ showed encouraging clinical results, with a mean Oxford Hip Score of 45.0 and a mean University of California at Los Angeles (UCLA) activity score of 7.4.

Similar to the prior increase in popularity of HRA, hip arthroscopy has also become much more commonplace, and its indications continue to evolve.¹¹ Hip arthroscopy has been used in the native hip joint to manage femoroacetabular impingement, labral tears, and iliopsoas tendinopathy, among other conditions.¹² In addition, the use of hip arthroscopy has not been limited to the native hip but also has increased as a diagnostic and therapeutic procedure after hip arthroplasties. Bajwa and Villar¹² found hip arthroscopy to be diagnostic in 23 of 24 patients who underwent the procedure after a hip arthroplasty, concluding that arthroscopy is a useful adjunct in the diagnosis of symptomatic arthroplasties.

Therefore, hip arthroscopy has been shown to be an effective modality to treat pathology in both the native hip and after hip arthroplasties. However, the effect of a prior hip arthroscopy on the outcome of a subsequent metal-on-metal HRA has not been determined. Piedade and colleagues¹³ showed a prior knee arthroscopy to increase the risk of postoperative complications and subsequent revision after total knee arthroplasty. Complications included reflex sympathetic dystrophy, undiagnosed pain, infection, stiffness, and component loosening. A prior osteochondroplasty at the femoral head-neck junction could increase the risk of femoral neck fracture after a subsequent HRA. Thus, the purpose of this study was to evaluate the clinical outcomes of a series of patients who received an HRA after a prior hip arthroscopy and to compare these results with a cohort of patients who received an HRA with no prior hip surgeries. Our hypothesis is that a prior hip arthroscopy will lead to inferior outcomes in patients undergoing HRA. 

Materials and Methods

This study is a retrospective, case-control study using a 1:2 matching analysis. Dr. Su performed all HRAs, which were enrolled in an institutional review board–approved arthroplasty registry. All HRAs were performed using the BHR System. 

The surgical technique for hip resurfacing arthroplasty has been described.¹ All procedures were performed via a posterior approach with the patient in the lateral decubitus position. All patients received a hybrid metal-on-metal hip resurfacing, with an uncemented acetabular component and cemented femoral component. Intraoperative anesthesia for all patients was performed via a combined spinal-epidural anesthetic, and an epidural patient-controlled analgesic was used for the first day postoperatively, followed by a transition to oral analgesics. The sizes of the acetabular and femoral components were recorded for each hip resurfacing. Postoperatively, intermittent pneumatic compression devices were placed upon arrival in the recovery room, and active ankle flexion and extension exercises were initiated immediately after the patient’s neurologic function returned.¹⁴ Aspirin was used for chemical deep venous thrombosis prophylaxis in all patients postoperatively for a period of 6 weeks. Full weight-bearing, with the use of crutches for assistance with balance, was permitted immediately. Crutches were used for a period of 3 weeks prior to being discontinued. 

From a database of 1357 HRAs (all BHR implants) performed between June 2006 and June 2012, 51 patients were identified who received an HRA after a prior hip arthroscopy. Eight patients were excluded because they did not possess adequate clinical documentation or were lost to follow-up. In the remaining 43 patients, there were 32 men and 11 women (21 right hips, 22 left hips), which formed the arthroscopy cohort. Two patients had a history of multiple hip arthroscopies (1 patient with 2 prior procedures, 1 patient with 3 prior procedures). The mean (SD) time from the most recent hip arthroscopy to the HRA was 2.5 (2.5) years. Table 1 presents a summary of the hip arthroscopy procedures (including only the most recent hip arthroscopy procedure in those with multiple arthroscopies).

Patient demographic variables (age, body mass index [BMI]) were recorded preoperatively, along with the Harris Hip Score (HHS),¹⁵ UCLA activity score,¹⁶ Western Ontario and McMaster Universities Osteoarthritis Index (WOMAC) score,¹⁷ and preoperative hip range of motion (flexion, extension, abduction, adduction, internal rotation, and external rotation). The same clinical indices were assessed postoperatively along with the Short Form-12 (SF-12) Health Survey Score,¹⁸ at the 6-week, 3-month, 6-month, 1-year, and most recent follow-up visits.

Radiographic assessment consisted of a low anteroposterior (AP) pelvic radiograph (with the radiographic beam centered on the pubic symphysis) and a cross-table lateral radiograph obtained at the most recent follow-up visit. Both the acetabular component abduction relative to the inter-teardrop line, and the angle between the femoral stem and the anatomic axis of the femoral shaft (stem-shaft angle) were measured on AP radiographs.^19,20 Acetabular component anteversion was measured on the cross-table lateral radiographs as the angle between the projected long axis of the acetabular opening and a line drawn perpendicular to the long axis plane of the body (Figures A, B).²¹

The same registry database was used to identify patients who received an HRA without a prior history of arthroscopy or hip surgery. A 1:2 matching analysis for those patients with a prior hip arthroscopy to those without a prior hip arthroscopy was performed to formulate a control group (control cohort) of 86 patients. Each patient in the arthroscopy cohort was matched with 2 patients in the control cohort based on the following parameters: age (± 6 years), sex (same), BMI (± 4 kg/m²), femoral head size (± 4 mm), and preoperative HHS and WOMAC scores (± 7 points). In the event an arthroscopy patient matched to 2 or more control patients, the patients who minimized the least squared error among the matching variables were selected. 

Statistical Analysis

All data were collected and analyzed using Microsoft Excel software (Microsoft Corporation, Redmond, Washington). Statistical comparisons between the 2 cohorts regarding demographic variables, clinical outcomes, and radiographic alignment were performed using an unpaired, Student 2-tailed t test, with statistical significance set at P ≤ .05. 

Results

A comparison of the results of the 1:2 matching analysis between the arthroscopy and control cohorts is presented in Table 2. There was no significant difference in the preoperative age, BMI, femoral head size, HHS, or WOMAC score between the 2 cohorts. However, the control cohort did show a more severe, preoperative flexion contracture (as expressed by a decreased amount of extension) and a decreased amount of preoperative abduction (Table 3). The preoperative UCLA activity score was also decreased in the control cohort, but this was not statistically significant.

The mean (SD) follow-up was 2.0 (1.0) years in the arthroscopy cohort and 2.1 (1.1) years in the control cohort. There was no significant difference in radiographic alignment between the 2 cohorts. The stem-shaft angle was 139.3° (SD, 5.4°) in the arthroscopy cohort (vs 138.3° [SD, 5.5°] in the control cohort; P = .3), the acetabular abduction was 43.9° (SD, 5.8°) in the arthroscopy cohort (vs 42.9° [SD, 6.1°] in the control cohort; P = .4), and the acetabular anteversion was 21.1° (SD, 7.5°) in the arthroscopy cohort (vs 20.8° [SD, 7.1°] in the control cohort; P = .8). 

At 6-week follow-up, the arthroscopy cohort showed a significantly decreased WOMAC score compared with the control cohort (72.9 [SD, 15.5] vs 80.5 [SD, 11.8], respectively; P = .05). In addition, there was a trend towards a decreased SF-12 mental component score in the arthroscopy cohort (52.2 [SD, 9.3] vs 56.5 [SD, 7.8] in the control cohort; P = .06). However, none of the remaining clinical indices showed a significant difference between the 2 cohorts, and there was no difference in range of motion between the 2 cohorts at the 6-week follow-up visit (Table 4).

In addition, at 3-month follow-up, no statistically significant differences were seen between the 2 cohorts for any of the clinical indices or range of motion values. Both groups continued to improve rapidly, with HHS of 96.9 (SD, 3.5) in the arthroscopy cohort and 95.5 (SD, 6.6) in the control cohort, and WOMAC scores of 88.7 (SD, 10.2) and 89.5 (SD, 9.8), respectively (Table 5). Similarly, at the 6-month and 1-year follow-up intervals, the 2 cohorts showed continued improvement in their clinical measures, with no statistically significant differences between the 2 cohorts (Tables 6, 7). 

At the most recent follow-up visit, more than 1 year after surgery, the HHS was 99.5 (SD, 1.3) in the arthroscopy cohort and 99.2 (SD, 9.7) in the control cohort (P = .9), and the WOMAC score was 93.5 (SD, 11.3) and 92.4 (SD, 12.2), respectively (P = .8). No significant perioperative complications were seen in the arthroscopy cohort. In the arthroscopy cohort, 1 patient was diagnosed with a deep venous thrombosis 2 weeks after the procedure and was placed on low-molecular-weight heparin and coumadin for treatment. A second patient in the arthroscopy cohort had continued serosanguinous drainage for 4 days postoperatively, which resolved with continued compressive dressings. To date, no patients in the arthroscopy or control cohorts have required a second operation or revision of their components.

Discussion

Given the increasing prevalence of hip arthroscopies to treat multiple disorders of the native joint, it is important to assess the potential consequences of these procedures on future arthroplasties. Piedade and colleagues,¹³ in a retrospective review of 1474 primary total knee arthroplasties, showed a prior bony procedure (high tibial osteotomy, tibial plateau fracture, patellar realignment) to be predictive of decreased range of motion postoperatively. In addition, a prior knee arthroscopy was associated with a higher rate of postoperative complications, with 30% of the complications requiring a reoperation, and 8.3% of the complications requiring a revision total knee arthroplasty. Kaplan-Meier survival curves showed a survival rate of only 86.8% in those patients with a prior knee arthroscopy (vs 98.1% in those without a prior knee surgery).²² Therefore, the purpose of this study was to evaluate the clinical outcomes of a series of patients who received an HRA after a prior hip arthroscopy. After the initial 6-week follow-up visit, no significant difference was seen in the functional outcomes between those patients with or without a history of prior hip arthroscopy who received an HRA.

After analysis of patient outcomes using multiple clinical measurement tools, at 6-week, 3-month, 6-month, 1-year, and most recent follow-up intervals, the only significant difference between the 2 cohorts was the WOMAC score at 6-week follow-up. Interestingly, there was no significant difference seen in the other clinical assessments, including the SF-12 score, HHS, range of motion, or UCLA activity score (although this did trend towards significance). This can be explained by the difference in both the mode of administration and various metrics assessed by these instruments. In comparison to the HHS evaluation, the patient completes the WOMAC (rather than the clinician) and also provides a more detailed assessment of symptoms, pain, stiffness, and activities of daily living.¹⁷ Therefore, this study suggests that patients with a prior hip arthroscopy may require more time to return to their activities of daily living after an HRA. However, whether the statistically significant difference between the 2 scores translates into a clinically significant difference can be questioned.

The clinical outcomes of this series of patients were excellent at the short-term follow-up, and both groups achieved clinical results comparable to prior reported results of HRA.^1,10,23,24 However, despite these results, there are several limitations to this study. First, longer-term follow-up is required to determine if any significant differences (such as aseptic loosening, infection, and prosthesis survival) are associated with a prior hip arthroscopy. In addition, this study included a relatively small cohort of patients who had a prior hip arthroscopy. However, a relatively large, single-surgeon database of 1357 HRAs was reviewed, with only 51 cases being reported (3.7%). With the increasing popularity of hip arthroscopy, the number of patients presenting for HRA will likely continue to increase. However, despite these limitations, this study shows that a prior hip arthroscopy does not appear to affect the short-term, clinical outcomes of a metal-on-metal HRA.

Metal-on-metal hip resurfacing arthroplasty (HRA) remains an alternative to total hip arthroplasty (THA) in appropriately selected, younger, active adults with degenerative hip disease.^1-4 While concerns remain regarding the potential for adverse local tissue reactions from wear of the metal-on-metal bearing surface,^5-8 10-year data from the Australian Orthopaedic Association National Joint Replacement Registry Annual Report⁹ showed a revision rate of only 6.3% when the Birmingham Hip Resurfacing (BHR) System was used (Smith & Nephew Inc, Memphis, Tennessee).In addition, in an independent review of 230 consecutive BHRs at a mean follow-up of 10.4 years, Coulter and colleagues¹⁰ showed encouraging clinical results, with a mean Oxford Hip Score of 45.0 and a mean University of California at Los Angeles (UCLA) activity score of 7.4.

Similar to the prior increase in popularity of HRA, hip arthroscopy has also become much more commonplace, and its indications continue to evolve.¹¹ Hip arthroscopy has been used in the native hip joint to manage femoroacetabular impingement, labral tears, and iliopsoas tendinopathy, among other conditions.¹² In addition, the use of hip arthroscopy has not been limited to the native hip but also has increased as a diagnostic and therapeutic procedure after hip arthroplasties. Bajwa and Villar¹² found hip arthroscopy to be diagnostic in 23 of 24 patients who underwent the procedure after a hip arthroplasty, concluding that arthroscopy is a useful adjunct in the diagnosis of symptomatic arthroplasties.

Therefore, hip arthroscopy has been shown to be an effective modality to treat pathology in both the native hip and after hip arthroplasties. However, the effect of a prior hip arthroscopy on the outcome of a subsequent metal-on-metal HRA has not been determined. Piedade and colleagues¹³ showed a prior knee arthroscopy to increase the risk of postoperative complications and subsequent revision after total knee arthroplasty. Complications included reflex sympathetic dystrophy, undiagnosed pain, infection, stiffness, and component loosening. A prior osteochondroplasty at the femoral head-neck junction could increase the risk of femoral neck fracture after a subsequent HRA. Thus, the purpose of this study was to evaluate the clinical outcomes of a series of patients who received an HRA after a prior hip arthroscopy and to compare these results with a cohort of patients who received an HRA with no prior hip surgeries. Our hypothesis is that a prior hip arthroscopy will lead to inferior outcomes in patients undergoing HRA. 

Materials and Methods

This study is a retrospective, case-control study using a 1:2 matching analysis. Dr. Su performed all HRAs, which were enrolled in an institutional review board–approved arthroplasty registry. All HRAs were performed using the BHR System. 

The surgical technique for hip resurfacing arthroplasty has been described.¹ All procedures were performed via a posterior approach with the patient in the lateral decubitus position. All patients received a hybrid metal-on-metal hip resurfacing, with an uncemented acetabular component and cemented femoral component. Intraoperative anesthesia for all patients was performed via a combined spinal-epidural anesthetic, and an epidural patient-controlled analgesic was used for the first day postoperatively, followed by a transition to oral analgesics. The sizes of the acetabular and femoral components were recorded for each hip resurfacing. Postoperatively, intermittent pneumatic compression devices were placed upon arrival in the recovery room, and active ankle flexion and extension exercises were initiated immediately after the patient’s neurologic function returned.¹⁴ Aspirin was used for chemical deep venous thrombosis prophylaxis in all patients postoperatively for a period of 6 weeks. Full weight-bearing, with the use of crutches for assistance with balance, was permitted immediately. Crutches were used for a period of 3 weeks prior to being discontinued. 

From a database of 1357 HRAs (all BHR implants) performed between June 2006 and June 2012, 51 patients were identified who received an HRA after a prior hip arthroscopy. Eight patients were excluded because they did not possess adequate clinical documentation or were lost to follow-up. In the remaining 43 patients, there were 32 men and 11 women (21 right hips, 22 left hips), which formed the arthroscopy cohort. Two patients had a history of multiple hip arthroscopies (1 patient with 2 prior procedures, 1 patient with 3 prior procedures). The mean (SD) time from the most recent hip arthroscopy to the HRA was 2.5 (2.5) years. Table 1 presents a summary of the hip arthroscopy procedures (including only the most recent hip arthroscopy procedure in those with multiple arthroscopies).

Patient demographic variables (age, body mass index [BMI]) were recorded preoperatively, along with the Harris Hip Score (HHS),¹⁵ UCLA activity score,¹⁶ Western Ontario and McMaster Universities Osteoarthritis Index (WOMAC) score,¹⁷ and preoperative hip range of motion (flexion, extension, abduction, adduction, internal rotation, and external rotation). The same clinical indices were assessed postoperatively along with the Short Form-12 (SF-12) Health Survey Score,¹⁸ at the 6-week, 3-month, 6-month, 1-year, and most recent follow-up visits.

Radiographic assessment consisted of a low anteroposterior (AP) pelvic radiograph (with the radiographic beam centered on the pubic symphysis) and a cross-table lateral radiograph obtained at the most recent follow-up visit. Both the acetabular component abduction relative to the inter-teardrop line, and the angle between the femoral stem and the anatomic axis of the femoral shaft (stem-shaft angle) were measured on AP radiographs.^19,20 Acetabular component anteversion was measured on the cross-table lateral radiographs as the angle between the projected long axis of the acetabular opening and a line drawn perpendicular to the long axis plane of the body (Figures A, B).²¹

The same registry database was used to identify patients who received an HRA without a prior history of arthroscopy or hip surgery. A 1:2 matching analysis for those patients with a prior hip arthroscopy to those without a prior hip arthroscopy was performed to formulate a control group (control cohort) of 86 patients. Each patient in the arthroscopy cohort was matched with 2 patients in the control cohort based on the following parameters: age (± 6 years), sex (same), BMI (± 4 kg/m²), femoral head size (± 4 mm), and preoperative HHS and WOMAC scores (± 7 points). In the event an arthroscopy patient matched to 2 or more control patients, the patients who minimized the least squared error among the matching variables were selected. 

Statistical Analysis

All data were collected and analyzed using Microsoft Excel software (Microsoft Corporation, Redmond, Washington). Statistical comparisons between the 2 cohorts regarding demographic variables, clinical outcomes, and radiographic alignment were performed using an unpaired, Student 2-tailed t test, with statistical significance set at P ≤ .05. 

Results

A comparison of the results of the 1:2 matching analysis between the arthroscopy and control cohorts is presented in Table 2. There was no significant difference in the preoperative age, BMI, femoral head size, HHS, or WOMAC score between the 2 cohorts. However, the control cohort did show a more severe, preoperative flexion contracture (as expressed by a decreased amount of extension) and a decreased amount of preoperative abduction (Table 3). The preoperative UCLA activity score was also decreased in the control cohort, but this was not statistically significant.

The mean (SD) follow-up was 2.0 (1.0) years in the arthroscopy cohort and 2.1 (1.1) years in the control cohort. There was no significant difference in radiographic alignment between the 2 cohorts. The stem-shaft angle was 139.3° (SD, 5.4°) in the arthroscopy cohort (vs 138.3° [SD, 5.5°] in the control cohort; P = .3), the acetabular abduction was 43.9° (SD, 5.8°) in the arthroscopy cohort (vs 42.9° [SD, 6.1°] in the control cohort; P = .4), and the acetabular anteversion was 21.1° (SD, 7.5°) in the arthroscopy cohort (vs 20.8° [SD, 7.1°] in the control cohort; P = .8). 

At 6-week follow-up, the arthroscopy cohort showed a significantly decreased WOMAC score compared with the control cohort (72.9 [SD, 15.5] vs 80.5 [SD, 11.8], respectively; P = .05). In addition, there was a trend towards a decreased SF-12 mental component score in the arthroscopy cohort (52.2 [SD, 9.3] vs 56.5 [SD, 7.8] in the control cohort; P = .06). However, none of the remaining clinical indices showed a significant difference between the 2 cohorts, and there was no difference in range of motion between the 2 cohorts at the 6-week follow-up visit (Table 4).

In addition, at 3-month follow-up, no statistically significant differences were seen between the 2 cohorts for any of the clinical indices or range of motion values. Both groups continued to improve rapidly, with HHS of 96.9 (SD, 3.5) in the arthroscopy cohort and 95.5 (SD, 6.6) in the control cohort, and WOMAC scores of 88.7 (SD, 10.2) and 89.5 (SD, 9.8), respectively (Table 5). Similarly, at the 6-month and 1-year follow-up intervals, the 2 cohorts showed continued improvement in their clinical measures, with no statistically significant differences between the 2 cohorts (Tables 6, 7). 

At the most recent follow-up visit, more than 1 year after surgery, the HHS was 99.5 (SD, 1.3) in the arthroscopy cohort and 99.2 (SD, 9.7) in the control cohort (P = .9), and the WOMAC score was 93.5 (SD, 11.3) and 92.4 (SD, 12.2), respectively (P = .8). No significant perioperative complications were seen in the arthroscopy cohort. In the arthroscopy cohort, 1 patient was diagnosed with a deep venous thrombosis 2 weeks after the procedure and was placed on low-molecular-weight heparin and coumadin for treatment. A second patient in the arthroscopy cohort had continued serosanguinous drainage for 4 days postoperatively, which resolved with continued compressive dressings. To date, no patients in the arthroscopy or control cohorts have required a second operation or revision of their components.

Discussion

Given the increasing prevalence of hip arthroscopies to treat multiple disorders of the native joint, it is important to assess the potential consequences of these procedures on future arthroplasties. Piedade and colleagues,¹³ in a retrospective review of 1474 primary total knee arthroplasties, showed a prior bony procedure (high tibial osteotomy, tibial plateau fracture, patellar realignment) to be predictive of decreased range of motion postoperatively. In addition, a prior knee arthroscopy was associated with a higher rate of postoperative complications, with 30% of the complications requiring a reoperation, and 8.3% of the complications requiring a revision total knee arthroplasty. Kaplan-Meier survival curves showed a survival rate of only 86.8% in those patients with a prior knee arthroscopy (vs 98.1% in those without a prior knee surgery).²² Therefore, the purpose of this study was to evaluate the clinical outcomes of a series of patients who received an HRA after a prior hip arthroscopy. After the initial 6-week follow-up visit, no significant difference was seen in the functional outcomes between those patients with or without a history of prior hip arthroscopy who received an HRA.

After analysis of patient outcomes using multiple clinical measurement tools, at 6-week, 3-month, 6-month, 1-year, and most recent follow-up intervals, the only significant difference between the 2 cohorts was the WOMAC score at 6-week follow-up. Interestingly, there was no significant difference seen in the other clinical assessments, including the SF-12 score, HHS, range of motion, or UCLA activity score (although this did trend towards significance). This can be explained by the difference in both the mode of administration and various metrics assessed by these instruments. In comparison to the HHS evaluation, the patient completes the WOMAC (rather than the clinician) and also provides a more detailed assessment of symptoms, pain, stiffness, and activities of daily living.¹⁷ Therefore, this study suggests that patients with a prior hip arthroscopy may require more time to return to their activities of daily living after an HRA. However, whether the statistically significant difference between the 2 scores translates into a clinically significant difference can be questioned.

The clinical outcomes of this series of patients were excellent at the short-term follow-up, and both groups achieved clinical results comparable to prior reported results of HRA.^1,10,23,24 However, despite these results, there are several limitations to this study. First, longer-term follow-up is required to determine if any significant differences (such as aseptic loosening, infection, and prosthesis survival) are associated with a prior hip arthroscopy. In addition, this study included a relatively small cohort of patients who had a prior hip arthroscopy. However, a relatively large, single-surgeon database of 1357 HRAs was reviewed, with only 51 cases being reported (3.7%). With the increasing popularity of hip arthroscopy, the number of patients presenting for HRA will likely continue to increase. However, despite these limitations, this study shows that a prior hip arthroscopy does not appear to affect the short-term, clinical outcomes of a metal-on-metal HRA.

Metal-on-metal hip resurfacing arthroplasty (HRA) remains an alternative to total hip arthroplasty (THA) in appropriately selected, younger, active adults with degenerative hip disease.^1-4 While concerns remain regarding the potential for adverse local tissue reactions from wear of the metal-on-metal bearing surface,^5-8 10-year data from the Australian Orthopaedic Association National Joint Replacement Registry Annual Report⁹ showed a revision rate of only 6.3% when the Birmingham Hip Resurfacing (BHR) System was used (Smith & Nephew Inc, Memphis, Tennessee).In addition, in an independent review of 230 consecutive BHRs at a mean follow-up of 10.4 years, Coulter and colleagues¹⁰ showed encouraging clinical results, with a mean Oxford Hip Score of 45.0 and a mean University of California at Los Angeles (UCLA) activity score of 7.4.

Similar to the prior increase in popularity of HRA, hip arthroscopy has also become much more commonplace, and its indications continue to evolve.¹¹ Hip arthroscopy has been used in the native hip joint to manage femoroacetabular impingement, labral tears, and iliopsoas tendinopathy, among other conditions.¹² In addition, the use of hip arthroscopy has not been limited to the native hip but also has increased as a diagnostic and therapeutic procedure after hip arthroplasties. Bajwa and Villar¹² found hip arthroscopy to be diagnostic in 23 of 24 patients who underwent the procedure after a hip arthroplasty, concluding that arthroscopy is a useful adjunct in the diagnosis of symptomatic arthroplasties.

Therefore, hip arthroscopy has been shown to be an effective modality to treat pathology in both the native hip and after hip arthroplasties. However, the effect of a prior hip arthroscopy on the outcome of a subsequent metal-on-metal HRA has not been determined. Piedade and colleagues¹³ showed a prior knee arthroscopy to increase the risk of postoperative complications and subsequent revision after total knee arthroplasty. Complications included reflex sympathetic dystrophy, undiagnosed pain, infection, stiffness, and component loosening. A prior osteochondroplasty at the femoral head-neck junction could increase the risk of femoral neck fracture after a subsequent HRA. Thus, the purpose of this study was to evaluate the clinical outcomes of a series of patients who received an HRA after a prior hip arthroscopy and to compare these results with a cohort of patients who received an HRA with no prior hip surgeries. Our hypothesis is that a prior hip arthroscopy will lead to inferior outcomes in patients undergoing HRA. 

Materials and Methods

This study is a retrospective, case-control study using a 1:2 matching analysis. Dr. Su performed all HRAs, which were enrolled in an institutional review board–approved arthroplasty registry. All HRAs were performed using the BHR System. 

The surgical technique for hip resurfacing arthroplasty has been described.¹ All procedures were performed via a posterior approach with the patient in the lateral decubitus position. All patients received a hybrid metal-on-metal hip resurfacing, with an uncemented acetabular component and cemented femoral component. Intraoperative anesthesia for all patients was performed via a combined spinal-epidural anesthetic, and an epidural patient-controlled analgesic was used for the first day postoperatively, followed by a transition to oral analgesics. The sizes of the acetabular and femoral components were recorded for each hip resurfacing. Postoperatively, intermittent pneumatic compression devices were placed upon arrival in the recovery room, and active ankle flexion and extension exercises were initiated immediately after the patient’s neurologic function returned.¹⁴ Aspirin was used for chemical deep venous thrombosis prophylaxis in all patients postoperatively for a period of 6 weeks. Full weight-bearing, with the use of crutches for assistance with balance, was permitted immediately. Crutches were used for a period of 3 weeks prior to being discontinued. 

From a database of 1357 HRAs (all BHR implants) performed between June 2006 and June 2012, 51 patients were identified who received an HRA after a prior hip arthroscopy. Eight patients were excluded because they did not possess adequate clinical documentation or were lost to follow-up. In the remaining 43 patients, there were 32 men and 11 women (21 right hips, 22 left hips), which formed the arthroscopy cohort. Two patients had a history of multiple hip arthroscopies (1 patient with 2 prior procedures, 1 patient with 3 prior procedures). The mean (SD) time from the most recent hip arthroscopy to the HRA was 2.5 (2.5) years. Table 1 presents a summary of the hip arthroscopy procedures (including only the most recent hip arthroscopy procedure in those with multiple arthroscopies).

Patient demographic variables (age, body mass index [BMI]) were recorded preoperatively, along with the Harris Hip Score (HHS),¹⁵ UCLA activity score,¹⁶ Western Ontario and McMaster Universities Osteoarthritis Index (WOMAC) score,¹⁷ and preoperative hip range of motion (flexion, extension, abduction, adduction, internal rotation, and external rotation). The same clinical indices were assessed postoperatively along with the Short Form-12 (SF-12) Health Survey Score,¹⁸ at the 6-week, 3-month, 6-month, 1-year, and most recent follow-up visits.

Radiographic assessment consisted of a low anteroposterior (AP) pelvic radiograph (with the radiographic beam centered on the pubic symphysis) and a cross-table lateral radiograph obtained at the most recent follow-up visit. Both the acetabular component abduction relative to the inter-teardrop line, and the angle between the femoral stem and the anatomic axis of the femoral shaft (stem-shaft angle) were measured on AP radiographs.^19,20 Acetabular component anteversion was measured on the cross-table lateral radiographs as the angle between the projected long axis of the acetabular opening and a line drawn perpendicular to the long axis plane of the body (Figures A, B).²¹

The same registry database was used to identify patients who received an HRA without a prior history of arthroscopy or hip surgery. A 1:2 matching analysis for those patients with a prior hip arthroscopy to those without a prior hip arthroscopy was performed to formulate a control group (control cohort) of 86 patients. Each patient in the arthroscopy cohort was matched with 2 patients in the control cohort based on the following parameters: age (± 6 years), sex (same), BMI (± 4 kg/m²), femoral head size (± 4 mm), and preoperative HHS and WOMAC scores (± 7 points). In the event an arthroscopy patient matched to 2 or more control patients, the patients who minimized the least squared error among the matching variables were selected. 

Statistical Analysis

All data were collected and analyzed using Microsoft Excel software (Microsoft Corporation, Redmond, Washington). Statistical comparisons between the 2 cohorts regarding demographic variables, clinical outcomes, and radiographic alignment were performed using an unpaired, Student 2-tailed t test, with statistical significance set at P ≤ .05. 

Results

A comparison of the results of the 1:2 matching analysis between the arthroscopy and control cohorts is presented in Table 2. There was no significant difference in the preoperative age, BMI, femoral head size, HHS, or WOMAC score between the 2 cohorts. However, the control cohort did show a more severe, preoperative flexion contracture (as expressed by a decreased amount of extension) and a decreased amount of preoperative abduction (Table 3). The preoperative UCLA activity score was also decreased in the control cohort, but this was not statistically significant.

The mean (SD) follow-up was 2.0 (1.0) years in the arthroscopy cohort and 2.1 (1.1) years in the control cohort. There was no significant difference in radiographic alignment between the 2 cohorts. The stem-shaft angle was 139.3° (SD, 5.4°) in the arthroscopy cohort (vs 138.3° [SD, 5.5°] in the control cohort; P = .3), the acetabular abduction was 43.9° (SD, 5.8°) in the arthroscopy cohort (vs 42.9° [SD, 6.1°] in the control cohort; P = .4), and the acetabular anteversion was 21.1° (SD, 7.5°) in the arthroscopy cohort (vs 20.8° [SD, 7.1°] in the control cohort; P = .8). 

At 6-week follow-up, the arthroscopy cohort showed a significantly decreased WOMAC score compared with the control cohort (72.9 [SD, 15.5] vs 80.5 [SD, 11.8], respectively; P = .05). In addition, there was a trend towards a decreased SF-12 mental component score in the arthroscopy cohort (52.2 [SD, 9.3] vs 56.5 [SD, 7.8] in the control cohort; P = .06). However, none of the remaining clinical indices showed a significant difference between the 2 cohorts, and there was no difference in range of motion between the 2 cohorts at the 6-week follow-up visit (Table 4).

In addition, at 3-month follow-up, no statistically significant differences were seen between the 2 cohorts for any of the clinical indices or range of motion values. Both groups continued to improve rapidly, with HHS of 96.9 (SD, 3.5) in the arthroscopy cohort and 95.5 (SD, 6.6) in the control cohort, and WOMAC scores of 88.7 (SD, 10.2) and 89.5 (SD, 9.8), respectively (Table 5). Similarly, at the 6-month and 1-year follow-up intervals, the 2 cohorts showed continued improvement in their clinical measures, with no statistically significant differences between the 2 cohorts (Tables 6, 7). 

At the most recent follow-up visit, more than 1 year after surgery, the HHS was 99.5 (SD, 1.3) in the arthroscopy cohort and 99.2 (SD, 9.7) in the control cohort (P = .9), and the WOMAC score was 93.5 (SD, 11.3) and 92.4 (SD, 12.2), respectively (P = .8). No significant perioperative complications were seen in the arthroscopy cohort. In the arthroscopy cohort, 1 patient was diagnosed with a deep venous thrombosis 2 weeks after the procedure and was placed on low-molecular-weight heparin and coumadin for treatment. A second patient in the arthroscopy cohort had continued serosanguinous drainage for 4 days postoperatively, which resolved with continued compressive dressings. To date, no patients in the arthroscopy or control cohorts have required a second operation or revision of their components.

Discussion

Given the increasing prevalence of hip arthroscopies to treat multiple disorders of the native joint, it is important to assess the potential consequences of these procedures on future arthroplasties. Piedade and colleagues,¹³ in a retrospective review of 1474 primary total knee arthroplasties, showed a prior bony procedure (high tibial osteotomy, tibial plateau fracture, patellar realignment) to be predictive of decreased range of motion postoperatively. In addition, a prior knee arthroscopy was associated with a higher rate of postoperative complications, with 30% of the complications requiring a reoperation, and 8.3% of the complications requiring a revision total knee arthroplasty. Kaplan-Meier survival curves showed a survival rate of only 86.8% in those patients with a prior knee arthroscopy (vs 98.1% in those without a prior knee surgery).²² Therefore, the purpose of this study was to evaluate the clinical outcomes of a series of patients who received an HRA after a prior hip arthroscopy. After the initial 6-week follow-up visit, no significant difference was seen in the functional outcomes between those patients with or without a history of prior hip arthroscopy who received an HRA.

After analysis of patient outcomes using multiple clinical measurement tools, at 6-week, 3-month, 6-month, 1-year, and most recent follow-up intervals, the only significant difference between the 2 cohorts was the WOMAC score at 6-week follow-up. Interestingly, there was no significant difference seen in the other clinical assessments, including the SF-12 score, HHS, range of motion, or UCLA activity score (although this did trend towards significance). This can be explained by the difference in both the mode of administration and various metrics assessed by these instruments. In comparison to the HHS evaluation, the patient completes the WOMAC (rather than the clinician) and also provides a more detailed assessment of symptoms, pain, stiffness, and activities of daily living.¹⁷ Therefore, this study suggests that patients with a prior hip arthroscopy may require more time to return to their activities of daily living after an HRA. However, whether the statistically significant difference between the 2 scores translates into a clinically significant difference can be questioned.

The clinical outcomes of this series of patients were excellent at the short-term follow-up, and both groups achieved clinical results comparable to prior reported results of HRA.^1,10,23,24 However, despite these results, there are several limitations to this study. First, longer-term follow-up is required to determine if any significant differences (such as aseptic loosening, infection, and prosthesis survival) are associated with a prior hip arthroscopy. In addition, this study included a relatively small cohort of patients who had a prior hip arthroscopy. However, a relatively large, single-surgeon database of 1357 HRAs was reviewed, with only 51 cases being reported (3.7%). With the increasing popularity of hip arthroscopy, the number of patients presenting for HRA will likely continue to increase. However, despite these limitations, this study shows that a prior hip arthroscopy does not appear to affect the short-term, clinical outcomes of a metal-on-metal HRA.

ANSWER
The radiograph demonstrates innumerable small lytic defects throughout the calvarium. The patient’s confusion is most likely secondary to profound metabolic abnormalities. However, in the setting of lytic bone lesions, metabolic abnormalities of renal insufficiency, severe hypercalcemia, and hypomagnesemia, one must be concerned about an occult myeloma, and appropriate work-up must be done.