Imaging

Synovial chondromatosis (SCM) is a relatively rare benign lesion of the synovium.¹ Its pathogenesis has been thought to be a chondral metaplasia of the subintimal layer of the intra- or extra-articular synovium.² However, evidence supporting a neoplastic cause of the disease is emerging.³ When intra-articular, any joint can be affected, though large joints are more prone to the disease; the knee, hip, and elbow are the most common locations.⁴ The synovial layer of tendons or bursae can be the origin of extra-articular SCM.⁵

Synovial chondrosarcoma (SCS), an even rarer pathology, can be caused by malignant transformation of SCM or can appear de novo on a synovial background.⁶ Histologic differentiation from SCM might be difficult because of the high incidence of hypercellularity, cellular atypia, and binucleated cells.⁶ Some features, such as presence of a very large mass or erosion of the surrounding bones, have been indicated as possible signs of malignancy.³ An unusual presentation of SCM, giant solitary synovial chondromatosis (GSSCM), can be hard to distinguish from SCS because of the large volume and possible aggressive radiologic findings.⁷ Some histologic features, such as presence of necrosis and mitotic cells, have been suggested as distinctive criteria for malignancy.⁸

In this article, we present a case of benign GSSCM with a histologic feature that has not been considered typical for benign SCM. The patient provided written informed consent for print and electronic publication of this case report.

Case Report

An 18-year-old woman presented with a large mass over the right hip. The mass had been growing slowly for 2 years. One year before presentation, a radiograph showed a large hip mass with fluffy calcification (Figure 1), and magnetic resonance imaging (MRI) showed a large nonhomogeneous mass anterior to the hip capsule and extending into the hip joint back to the posterior part of the joint (Figures 2A, 2B). Open incisional biopsy was performed in a local hospital at the time, and the histologic analysis revealed presence of atypical binucleated cells and pleomorphism, in addition to some mitotic activity (0 to 1 per high-power field) (Figure 3). These findings suggested malignancy. The patient declined surgery up until the time she presented to our hospital, 1 year later.

Clinical examination findings on admission to our hospital were striking. The patient had a large mass in the groin region. It was fairly tender and firm to palpation, immobile, and close to the skin. Hip motion was mildly painful but obviously restricted.

The mass was restaged. New radiographs and MRI did not show any significant changes since the previous year, computed tomography (CT) did not show any bone erosion (Figure 4), and chest radiograph, CT, and whole-body bone scan did not demonstrate any signs of metastasis.

Given the clinical presentation and previous histopathologic findings, a diagnosis of GSSCM with possible malignant transformation was made. The patient was scheduled for surgery. During surgery, the tumor was exposed through the Smith-Petersen approach. The mass was extruding under the fascia between the femoral neurovascular bundle medially and iliopsoas muscle laterally. There was no adhesion of the surrounding structures, including the femoral neurovascular bundle, to the mass. The muscle was sitting on the anterolateral surface of the mass, which was considered located in the iliopsoas bursa but extending to the joint. In the vertical plane, the mass extended down to the subtrochanteric area. The entire solid extra-articular mass was excised en bloc, and hip capsulotomy was performed inferior to the area of emergence of the mass. The joint was occupied by a single solid cartilaginous mass molding around the femoral neck, filling the piriformis fossa and propagating to the posterior joint space. Obtaining enough exposure to the back of the joint required surgical hip dislocation. The visualized acetabular fossa revealed chondral fragments, which were excised. Bone erosion or significant osteoarthritis was not detected in any part of the joint. A nearly total synovectomy was performed, leaving the ascending retinacular vessels intact. Meticulous technique was used to avoid contaminating the extra-articular tissues. The wound was closed in the routine way after hip relocation.

The 16×9.5×9-cm mass (Figure 5A) had a conglomerated internal structure (Figure 5B). Multiple specimens from the intra- and extra-articular portions of the mass were sent for histopathologic analysis, which revealed clusters of mature chondrocytes arranged in a lobular pattern and separated by thin fibrous bands. Areas of calcification and ossification were appreciated as well (Figures 6A-6C). No necrosis, mitosis, or bone permeation was detected. These findings were compatible with typical SCM. Given these pathologic findings and the lack of clinical deterioration over the previous year, a diagnosis of GSSCM with extension along the iliopsoas and obturator externus bursae was made. The already-performed marginal excision was deemed sufficient treatment. At most recent follow-up, 38 months after surgery, the patient was pain-free and had good hip range of motion and no indication of recurrence.

Discussion

SCM is a benign disorder emerging from the synovium as a result of proliferative changes in the synovial membrane of the joints, tendon sheaths, or bursae, leading to the formation of numerous cartilaginous nodules, usually a few millimeters in diameter.⁸ In a rare presentation of the disease, the nodules may coalesce to form a large mass, or a single cartilaginous nodule may enlarge to form a mass. Edeiken and colleagues⁷ named this previously unrecognized SCM feature as GSSCM when there was a major single mass larger than 1 cm in diameter. There have been other SCM cases with multiple giant masses.^9,10 In the English-language literature, we found 15 GSSCM cases, which include the first reported, by Edeiken and colleagues⁷ (Table). However, earlier SCM cases would be reclassified GSSCM according to their definition.¹¹

The present case brings the total to 16. Nine of the 16 patients were male. Mean age at presentation was 41 years (range, 10-80 years). The knee was the most common GSSCM site (6 cases), followed by the temporomandibular and hip joints (3 each). Regarding gross pathology, 10 lesions were solid, and 6 (including the present one) were formed by conglomeration of the chondromatosis nodules. Lesions varied in size (16-200 mm), and 2 were primarily extra-articular (foot). One common issue with most of the cases was the initial diagnosis of chondrosarcoma. The exact surgical technique used was described for 6 cases (cases 11-16); the technique was marginal excision. In no case was recurrence 14 to 60 months after surgery reported.

This chondroproliferative process is potentially a diagnostic challenge, as distinguishing it from a chondrosarcoma, a more common lesion, could be difficult based on clinical and imaging findings, and, as is true for other chondral lesions, even histologic differentiation of the conditions might not be conclusive.^12,13 Confusion in diagnosis was almost universal in this series of patients.

One important differentiating feature of benign and malignant skeletal lesions is the time course of the disease. Malignant tumors are expected to demonstrate rapid enlargement and local or systemic spread. Unfortunately, often SCS cannot be distinguished by this characteristic, as grade I or II chondrosarcoma is usually a slow-growing tumor and does not metastasize early.¹⁴ Although lack of recurrence is assuring, recurrence is not necessarily a sign of malignancy, as a considerable percentage of benign chondromatosis lesions recur.⁸

Radiologic differentiation between SCM and SCS is another challenge. Although bone erosion caused by a lesion not originating from bone is usually considered a sign of malignancy, GSSCM was reported as causing bone erosion in 5 of the 16 cases in our literature review.^7,15 Our patient did not experience any bone erosion. However, lack of bone erosion is not a reliable criterion for excluding SCS, and bone erosion was noted in only 3 of the 9 SCS cases in the series reported by Bertoni and colleagues.⁶ Moreover, tumor size and propagation of tumor to surrounding tissue could be surprising in GSSCM. Large size (up to 20 cm) and extra-articular spread of a lesion originating in a joint are common findings.^6,16 Our case was an obvious extension of a hip GSSCM to the iliopsoas and obturator externus bursa, which is the most common pattern of extracapsular spread of hip SCM.¹⁷ An interesting feature of the present case, however, was the relatively superficial location of the mass immediately under the fascia.

Calcified matrix is key in diagnosing a chondral lesion on imaging studies, but, in some cases, SCM does not demonstrate any radiographically detectable calcification at time of diagnosis.¹⁸ However, all the GSSCM cases reported to date had obvious calcified matrix.

The hypercellularity, cellular atypia, binucleated cells, and pleomorphism in the histologic examination of the present case are not features of malignancy in SCM.⁸ On the contrary, several other characteristics, including qualitative differences in the arrangement of chondrocytes (sheets rather than clusters), myxoid matrix, hypercellularity with crowding and spindling of the nuclei at the periphery, necrosis, and, most important, permeation of the trabecular bone with the filling up of marrow spaces, have been assumed to be indicative of malignancy.⁸ Furthermore, Davis and colleagues⁸ found no mitotic activity in the histopathologic investigation of 53 SCM cases. Even in 3 cases that developed malignant transformation to SCS, mitosis was not found in the initial biopsy specimens before transformation. This was compatible with the common opinion that SCM is not a neoplastic, but a metaplastic, process. Histopathologic data were available for only 8 of the previous 15 GSSCM cases. There were no reports of mitosis, and necrosis was found in only 1 case.¹⁶ In our patient’s case, however, the first biopsy did show remarkable mitotic activity. This was not the case for the second biopsy, when mature chondrocytes associated with marked calcification and ossification were prominent features (Figures 6A, 6B). We presume that, within a limited period during earlier stages of tissue maturation in SCM, mitotic activity might be a possible finding. Of note, none of the other aforementioned histologic criteria for malignancy was seen in the first or second biopsy in the present case (Figures 3, 6C).

The original idea that SCM originates from a metaplasia in the subintimal layer of the synovium, where the synovium is in direct contact with the articular cartilage, has been challenged. The high incidence of hypercellularity, binucleated cells, and cellular atypia was always an argument against a metaplastic origin for the disease. Evidence of clonal chromosomal changes, like translocation of chromosome 1218 and chromosome 5 and 6 abnormalities,^19,20 in addition to other alterations,^19,21 provide some evidence supporting a neoplastic rather than a metaplastic origin for SCM. Given the presence of mitosis in the present case, the lack of mitotic activity in SCM, as stated by other authors,²² is not a universal feature and cannot be used as an argument against a neoplastic origin for SCM.

Although mitotic activity is uncommon in SCM, the present case illustrates the possible presence of mitotic activity in GSSCM. The simple presence of mitotic activity, a common finding in some other chondral tumors,23,24 does not preclude the diagnosis of benign SCM, as suggested before,8 and correlation of the clinical and radiologic manifestations with histopathologic findings is crucial for a correct diagnosis.

Synovial chondromatosis (SCM) is a relatively rare benign lesion of the synovium.¹ Its pathogenesis has been thought to be a chondral metaplasia of the subintimal layer of the intra- or extra-articular synovium.² However, evidence supporting a neoplastic cause of the disease is emerging.³ When intra-articular, any joint can be affected, though large joints are more prone to the disease; the knee, hip, and elbow are the most common locations.⁴ The synovial layer of tendons or bursae can be the origin of extra-articular SCM.⁵

Synovial chondrosarcoma (SCS), an even rarer pathology, can be caused by malignant transformation of SCM or can appear de novo on a synovial background.⁶ Histologic differentiation from SCM might be difficult because of the high incidence of hypercellularity, cellular atypia, and binucleated cells.⁶ Some features, such as presence of a very large mass or erosion of the surrounding bones, have been indicated as possible signs of malignancy.³ An unusual presentation of SCM, giant solitary synovial chondromatosis (GSSCM), can be hard to distinguish from SCS because of the large volume and possible aggressive radiologic findings.⁷ Some histologic features, such as presence of necrosis and mitotic cells, have been suggested as distinctive criteria for malignancy.⁸

In this article, we present a case of benign GSSCM with a histologic feature that has not been considered typical for benign SCM. The patient provided written informed consent for print and electronic publication of this case report.

Case Report

An 18-year-old woman presented with a large mass over the right hip. The mass had been growing slowly for 2 years. One year before presentation, a radiograph showed a large hip mass with fluffy calcification (Figure 1), and magnetic resonance imaging (MRI) showed a large nonhomogeneous mass anterior to the hip capsule and extending into the hip joint back to the posterior part of the joint (Figures 2A, 2B). Open incisional biopsy was performed in a local hospital at the time, and the histologic analysis revealed presence of atypical binucleated cells and pleomorphism, in addition to some mitotic activity (0 to 1 per high-power field) (Figure 3). These findings suggested malignancy. The patient declined surgery up until the time she presented to our hospital, 1 year later.

Clinical examination findings on admission to our hospital were striking. The patient had a large mass in the groin region. It was fairly tender and firm to palpation, immobile, and close to the skin. Hip motion was mildly painful but obviously restricted.

The mass was restaged. New radiographs and MRI did not show any significant changes since the previous year, computed tomography (CT) did not show any bone erosion (Figure 4), and chest radiograph, CT, and whole-body bone scan did not demonstrate any signs of metastasis.

Given the clinical presentation and previous histopathologic findings, a diagnosis of GSSCM with possible malignant transformation was made. The patient was scheduled for surgery. During surgery, the tumor was exposed through the Smith-Petersen approach. The mass was extruding under the fascia between the femoral neurovascular bundle medially and iliopsoas muscle laterally. There was no adhesion of the surrounding structures, including the femoral neurovascular bundle, to the mass. The muscle was sitting on the anterolateral surface of the mass, which was considered located in the iliopsoas bursa but extending to the joint. In the vertical plane, the mass extended down to the subtrochanteric area. The entire solid extra-articular mass was excised en bloc, and hip capsulotomy was performed inferior to the area of emergence of the mass. The joint was occupied by a single solid cartilaginous mass molding around the femoral neck, filling the piriformis fossa and propagating to the posterior joint space. Obtaining enough exposure to the back of the joint required surgical hip dislocation. The visualized acetabular fossa revealed chondral fragments, which were excised. Bone erosion or significant osteoarthritis was not detected in any part of the joint. A nearly total synovectomy was performed, leaving the ascending retinacular vessels intact. Meticulous technique was used to avoid contaminating the extra-articular tissues. The wound was closed in the routine way after hip relocation.

The 16×9.5×9-cm mass (Figure 5A) had a conglomerated internal structure (Figure 5B). Multiple specimens from the intra- and extra-articular portions of the mass were sent for histopathologic analysis, which revealed clusters of mature chondrocytes arranged in a lobular pattern and separated by thin fibrous bands. Areas of calcification and ossification were appreciated as well (Figures 6A-6C). No necrosis, mitosis, or bone permeation was detected. These findings were compatible with typical SCM. Given these pathologic findings and the lack of clinical deterioration over the previous year, a diagnosis of GSSCM with extension along the iliopsoas and obturator externus bursae was made. The already-performed marginal excision was deemed sufficient treatment. At most recent follow-up, 38 months after surgery, the patient was pain-free and had good hip range of motion and no indication of recurrence.

Discussion

SCM is a benign disorder emerging from the synovium as a result of proliferative changes in the synovial membrane of the joints, tendon sheaths, or bursae, leading to the formation of numerous cartilaginous nodules, usually a few millimeters in diameter.⁸ In a rare presentation of the disease, the nodules may coalesce to form a large mass, or a single cartilaginous nodule may enlarge to form a mass. Edeiken and colleagues⁷ named this previously unrecognized SCM feature as GSSCM when there was a major single mass larger than 1 cm in diameter. There have been other SCM cases with multiple giant masses.^9,10 In the English-language literature, we found 15 GSSCM cases, which include the first reported, by Edeiken and colleagues⁷ (Table). However, earlier SCM cases would be reclassified GSSCM according to their definition.¹¹

The present case brings the total to 16. Nine of the 16 patients were male. Mean age at presentation was 41 years (range, 10-80 years). The knee was the most common GSSCM site (6 cases), followed by the temporomandibular and hip joints (3 each). Regarding gross pathology, 10 lesions were solid, and 6 (including the present one) were formed by conglomeration of the chondromatosis nodules. Lesions varied in size (16-200 mm), and 2 were primarily extra-articular (foot). One common issue with most of the cases was the initial diagnosis of chondrosarcoma. The exact surgical technique used was described for 6 cases (cases 11-16); the technique was marginal excision. In no case was recurrence 14 to 60 months after surgery reported.

This chondroproliferative process is potentially a diagnostic challenge, as distinguishing it from a chondrosarcoma, a more common lesion, could be difficult based on clinical and imaging findings, and, as is true for other chondral lesions, even histologic differentiation of the conditions might not be conclusive.^12,13 Confusion in diagnosis was almost universal in this series of patients.

One important differentiating feature of benign and malignant skeletal lesions is the time course of the disease. Malignant tumors are expected to demonstrate rapid enlargement and local or systemic spread. Unfortunately, often SCS cannot be distinguished by this characteristic, as grade I or II chondrosarcoma is usually a slow-growing tumor and does not metastasize early.¹⁴ Although lack of recurrence is assuring, recurrence is not necessarily a sign of malignancy, as a considerable percentage of benign chondromatosis lesions recur.⁸

Radiologic differentiation between SCM and SCS is another challenge. Although bone erosion caused by a lesion not originating from bone is usually considered a sign of malignancy, GSSCM was reported as causing bone erosion in 5 of the 16 cases in our literature review.^7,15 Our patient did not experience any bone erosion. However, lack of bone erosion is not a reliable criterion for excluding SCS, and bone erosion was noted in only 3 of the 9 SCS cases in the series reported by Bertoni and colleagues.⁶ Moreover, tumor size and propagation of tumor to surrounding tissue could be surprising in GSSCM. Large size (up to 20 cm) and extra-articular spread of a lesion originating in a joint are common findings.^6,16 Our case was an obvious extension of a hip GSSCM to the iliopsoas and obturator externus bursa, which is the most common pattern of extracapsular spread of hip SCM.¹⁷ An interesting feature of the present case, however, was the relatively superficial location of the mass immediately under the fascia.

Calcified matrix is key in diagnosing a chondral lesion on imaging studies, but, in some cases, SCM does not demonstrate any radiographically detectable calcification at time of diagnosis.¹⁸ However, all the GSSCM cases reported to date had obvious calcified matrix.

The hypercellularity, cellular atypia, binucleated cells, and pleomorphism in the histologic examination of the present case are not features of malignancy in SCM.⁸ On the contrary, several other characteristics, including qualitative differences in the arrangement of chondrocytes (sheets rather than clusters), myxoid matrix, hypercellularity with crowding and spindling of the nuclei at the periphery, necrosis, and, most important, permeation of the trabecular bone with the filling up of marrow spaces, have been assumed to be indicative of malignancy.⁸ Furthermore, Davis and colleagues⁸ found no mitotic activity in the histopathologic investigation of 53 SCM cases. Even in 3 cases that developed malignant transformation to SCS, mitosis was not found in the initial biopsy specimens before transformation. This was compatible with the common opinion that SCM is not a neoplastic, but a metaplastic, process. Histopathologic data were available for only 8 of the previous 15 GSSCM cases. There were no reports of mitosis, and necrosis was found in only 1 case.¹⁶ In our patient’s case, however, the first biopsy did show remarkable mitotic activity. This was not the case for the second biopsy, when mature chondrocytes associated with marked calcification and ossification were prominent features (Figures 6A, 6B). We presume that, within a limited period during earlier stages of tissue maturation in SCM, mitotic activity might be a possible finding. Of note, none of the other aforementioned histologic criteria for malignancy was seen in the first or second biopsy in the present case (Figures 3, 6C).

The original idea that SCM originates from a metaplasia in the subintimal layer of the synovium, where the synovium is in direct contact with the articular cartilage, has been challenged. The high incidence of hypercellularity, binucleated cells, and cellular atypia was always an argument against a metaplastic origin for the disease. Evidence of clonal chromosomal changes, like translocation of chromosome 1218 and chromosome 5 and 6 abnormalities,^19,20 in addition to other alterations,^19,21 provide some evidence supporting a neoplastic rather than a metaplastic origin for SCM. Given the presence of mitosis in the present case, the lack of mitotic activity in SCM, as stated by other authors,²² is not a universal feature and cannot be used as an argument against a neoplastic origin for SCM.

Although mitotic activity is uncommon in SCM, the present case illustrates the possible presence of mitotic activity in GSSCM. The simple presence of mitotic activity, a common finding in some other chondral tumors,23,24 does not preclude the diagnosis of benign SCM, as suggested before,8 and correlation of the clinical and radiologic manifestations with histopathologic findings is crucial for a correct diagnosis.

Synovial chondromatosis (SCM) is a relatively rare benign lesion of the synovium.¹ Its pathogenesis has been thought to be a chondral metaplasia of the subintimal layer of the intra- or extra-articular synovium.² However, evidence supporting a neoplastic cause of the disease is emerging.³ When intra-articular, any joint can be affected, though large joints are more prone to the disease; the knee, hip, and elbow are the most common locations.⁴ The synovial layer of tendons or bursae can be the origin of extra-articular SCM.⁵

Synovial chondrosarcoma (SCS), an even rarer pathology, can be caused by malignant transformation of SCM or can appear de novo on a synovial background.⁶ Histologic differentiation from SCM might be difficult because of the high incidence of hypercellularity, cellular atypia, and binucleated cells.⁶ Some features, such as presence of a very large mass or erosion of the surrounding bones, have been indicated as possible signs of malignancy.³ An unusual presentation of SCM, giant solitary synovial chondromatosis (GSSCM), can be hard to distinguish from SCS because of the large volume and possible aggressive radiologic findings.⁷ Some histologic features, such as presence of necrosis and mitotic cells, have been suggested as distinctive criteria for malignancy.⁸

In this article, we present a case of benign GSSCM with a histologic feature that has not been considered typical for benign SCM. The patient provided written informed consent for print and electronic publication of this case report.

Case Report

An 18-year-old woman presented with a large mass over the right hip. The mass had been growing slowly for 2 years. One year before presentation, a radiograph showed a large hip mass with fluffy calcification (Figure 1), and magnetic resonance imaging (MRI) showed a large nonhomogeneous mass anterior to the hip capsule and extending into the hip joint back to the posterior part of the joint (Figures 2A, 2B). Open incisional biopsy was performed in a local hospital at the time, and the histologic analysis revealed presence of atypical binucleated cells and pleomorphism, in addition to some mitotic activity (0 to 1 per high-power field) (Figure 3). These findings suggested malignancy. The patient declined surgery up until the time she presented to our hospital, 1 year later.

Clinical examination findings on admission to our hospital were striking. The patient had a large mass in the groin region. It was fairly tender and firm to palpation, immobile, and close to the skin. Hip motion was mildly painful but obviously restricted.

The mass was restaged. New radiographs and MRI did not show any significant changes since the previous year, computed tomography (CT) did not show any bone erosion (Figure 4), and chest radiograph, CT, and whole-body bone scan did not demonstrate any signs of metastasis.

Given the clinical presentation and previous histopathologic findings, a diagnosis of GSSCM with possible malignant transformation was made. The patient was scheduled for surgery. During surgery, the tumor was exposed through the Smith-Petersen approach. The mass was extruding under the fascia between the femoral neurovascular bundle medially and iliopsoas muscle laterally. There was no adhesion of the surrounding structures, including the femoral neurovascular bundle, to the mass. The muscle was sitting on the anterolateral surface of the mass, which was considered located in the iliopsoas bursa but extending to the joint. In the vertical plane, the mass extended down to the subtrochanteric area. The entire solid extra-articular mass was excised en bloc, and hip capsulotomy was performed inferior to the area of emergence of the mass. The joint was occupied by a single solid cartilaginous mass molding around the femoral neck, filling the piriformis fossa and propagating to the posterior joint space. Obtaining enough exposure to the back of the joint required surgical hip dislocation. The visualized acetabular fossa revealed chondral fragments, which were excised. Bone erosion or significant osteoarthritis was not detected in any part of the joint. A nearly total synovectomy was performed, leaving the ascending retinacular vessels intact. Meticulous technique was used to avoid contaminating the extra-articular tissues. The wound was closed in the routine way after hip relocation.

The 16×9.5×9-cm mass (Figure 5A) had a conglomerated internal structure (Figure 5B). Multiple specimens from the intra- and extra-articular portions of the mass were sent for histopathologic analysis, which revealed clusters of mature chondrocytes arranged in a lobular pattern and separated by thin fibrous bands. Areas of calcification and ossification were appreciated as well (Figures 6A-6C). No necrosis, mitosis, or bone permeation was detected. These findings were compatible with typical SCM. Given these pathologic findings and the lack of clinical deterioration over the previous year, a diagnosis of GSSCM with extension along the iliopsoas and obturator externus bursae was made. The already-performed marginal excision was deemed sufficient treatment. At most recent follow-up, 38 months after surgery, the patient was pain-free and had good hip range of motion and no indication of recurrence.

Discussion

SCM is a benign disorder emerging from the synovium as a result of proliferative changes in the synovial membrane of the joints, tendon sheaths, or bursae, leading to the formation of numerous cartilaginous nodules, usually a few millimeters in diameter.⁸ In a rare presentation of the disease, the nodules may coalesce to form a large mass, or a single cartilaginous nodule may enlarge to form a mass. Edeiken and colleagues⁷ named this previously unrecognized SCM feature as GSSCM when there was a major single mass larger than 1 cm in diameter. There have been other SCM cases with multiple giant masses.^9,10 In the English-language literature, we found 15 GSSCM cases, which include the first reported, by Edeiken and colleagues⁷ (Table). However, earlier SCM cases would be reclassified GSSCM according to their definition.¹¹

The present case brings the total to 16. Nine of the 16 patients were male. Mean age at presentation was 41 years (range, 10-80 years). The knee was the most common GSSCM site (6 cases), followed by the temporomandibular and hip joints (3 each). Regarding gross pathology, 10 lesions were solid, and 6 (including the present one) were formed by conglomeration of the chondromatosis nodules. Lesions varied in size (16-200 mm), and 2 were primarily extra-articular (foot). One common issue with most of the cases was the initial diagnosis of chondrosarcoma. The exact surgical technique used was described for 6 cases (cases 11-16); the technique was marginal excision. In no case was recurrence 14 to 60 months after surgery reported.

This chondroproliferative process is potentially a diagnostic challenge, as distinguishing it from a chondrosarcoma, a more common lesion, could be difficult based on clinical and imaging findings, and, as is true for other chondral lesions, even histologic differentiation of the conditions might not be conclusive.^12,13 Confusion in diagnosis was almost universal in this series of patients.

One important differentiating feature of benign and malignant skeletal lesions is the time course of the disease. Malignant tumors are expected to demonstrate rapid enlargement and local or systemic spread. Unfortunately, often SCS cannot be distinguished by this characteristic, as grade I or II chondrosarcoma is usually a slow-growing tumor and does not metastasize early.¹⁴ Although lack of recurrence is assuring, recurrence is not necessarily a sign of malignancy, as a considerable percentage of benign chondromatosis lesions recur.⁸

Radiologic differentiation between SCM and SCS is another challenge. Although bone erosion caused by a lesion not originating from bone is usually considered a sign of malignancy, GSSCM was reported as causing bone erosion in 5 of the 16 cases in our literature review.^7,15 Our patient did not experience any bone erosion. However, lack of bone erosion is not a reliable criterion for excluding SCS, and bone erosion was noted in only 3 of the 9 SCS cases in the series reported by Bertoni and colleagues.⁶ Moreover, tumor size and propagation of tumor to surrounding tissue could be surprising in GSSCM. Large size (up to 20 cm) and extra-articular spread of a lesion originating in a joint are common findings.^6,16 Our case was an obvious extension of a hip GSSCM to the iliopsoas and obturator externus bursa, which is the most common pattern of extracapsular spread of hip SCM.¹⁷ An interesting feature of the present case, however, was the relatively superficial location of the mass immediately under the fascia.

Calcified matrix is key in diagnosing a chondral lesion on imaging studies, but, in some cases, SCM does not demonstrate any radiographically detectable calcification at time of diagnosis.¹⁸ However, all the GSSCM cases reported to date had obvious calcified matrix.

The hypercellularity, cellular atypia, binucleated cells, and pleomorphism in the histologic examination of the present case are not features of malignancy in SCM.⁸ On the contrary, several other characteristics, including qualitative differences in the arrangement of chondrocytes (sheets rather than clusters), myxoid matrix, hypercellularity with crowding and spindling of the nuclei at the periphery, necrosis, and, most important, permeation of the trabecular bone with the filling up of marrow spaces, have been assumed to be indicative of malignancy.⁸ Furthermore, Davis and colleagues⁸ found no mitotic activity in the histopathologic investigation of 53 SCM cases. Even in 3 cases that developed malignant transformation to SCS, mitosis was not found in the initial biopsy specimens before transformation. This was compatible with the common opinion that SCM is not a neoplastic, but a metaplastic, process. Histopathologic data were available for only 8 of the previous 15 GSSCM cases. There were no reports of mitosis, and necrosis was found in only 1 case.¹⁶ In our patient’s case, however, the first biopsy did show remarkable mitotic activity. This was not the case for the second biopsy, when mature chondrocytes associated with marked calcification and ossification were prominent features (Figures 6A, 6B). We presume that, within a limited period during earlier stages of tissue maturation in SCM, mitotic activity might be a possible finding. Of note, none of the other aforementioned histologic criteria for malignancy was seen in the first or second biopsy in the present case (Figures 3, 6C).

The original idea that SCM originates from a metaplasia in the subintimal layer of the synovium, where the synovium is in direct contact with the articular cartilage, has been challenged. The high incidence of hypercellularity, binucleated cells, and cellular atypia was always an argument against a metaplastic origin for the disease. Evidence of clonal chromosomal changes, like translocation of chromosome 1218 and chromosome 5 and 6 abnormalities,^19,20 in addition to other alterations,^19,21 provide some evidence supporting a neoplastic rather than a metaplastic origin for SCM. Given the presence of mitosis in the present case, the lack of mitotic activity in SCM, as stated by other authors,²² is not a universal feature and cannot be used as an argument against a neoplastic origin for SCM.

Although mitotic activity is uncommon in SCM, the present case illustrates the possible presence of mitotic activity in GSSCM. The simple presence of mitotic activity, a common finding in some other chondral tumors,23,24 does not preclude the diagnosis of benign SCM, as suggested before,8 and correlation of the clinical and radiologic manifestations with histopathologic findings is crucial for a correct diagnosis.

Anterior hip dislocations have been reported to account for approximately 5% to 10% of all hip dislocations.¹ Epstein and Wiss² originally divided anterior hip dislocations into superior (type I, including pubic or subspinous) and inferior (type II, including obturator and perineal) dislocations. This classification was further subdivided based on the presence of either no associated fracture (type A), fracture of the femoral head or neck (FNF; type B), or fracture of the acetabulum (type C).³ Of all anterior hip dislocations, it has been reported that the inferior or obturator type of dislocation is more common, constituting approximately 70% of all anterior dislocations.⁴ In 1943, Pringle⁵ described the mechanism of obturator dislocation as simultaneous abduction, flexion, and external rotation of the hip. Our literature search found only 2 case reports in non-English-language journals of a complete FNF associated with an attempted reduction of an anterior hip dislocation.^6,7 Indentation fractures of the femoral head have been more commonly reported than FNFs, with a reported incidence of 35% to 55% after anterior dislocation.^4,8 DeLee and colleagues⁸ also found that those patients with indentation fractures were at a higher risk for developing avascular necrosis of the femoral head in addition to being more likely to report poor or fair function of the hip 2 years after reduction.

There have been a number of different reduction maneuvers for anterior dislocation of hips published in the literature. Epstein and Harvey⁹ advocated reduction by traction in the line of the femur with the hip flexed and in gentle internal rotation and abduction while the patient was under general anesthesia. Toms and Williams,¹⁰ however, recommended adduction with gradual release of the longitudinal traction. Polesky and Polesky¹¹ described a reduction method involving sharp internal rotation, which was found to be associated with FNF. The patient provided written informed consent for print and electronic publication of this case report, and approval was obtained from the Emory University Institutional Review Board.

Case Report

The patient was a 73-year-old woman, an independent ambulator with minimal antecedent hip pain, who, as a pedestrian, was struck by a heavy-duty pickup truck at low velocity. She was flown to our level I trauma center from an outside hospital. The patient arrived hemodynamically stable, with a Glasgow Coma Scale score of 15 and with major complaints of right shoulder and right hip pain. She had a positive Focused Assessment with Sonography for Trauma (FAST), and underwent a subsequent urgent chest, abdomen, and pelvis computed tomography (CT) scan for further investigation. CT showed a grade 1 liver laceration. Her anteroposterior (AP) pelvic radiograph and pelvic CT scan showed an anterior hip dislocation with the femoral head located adjacent to the obturator foramen (Figures 1, 2). The AP pelvic radiograph and pelvic CT scan were scrutinized extensively before reduction to rule out a possible FNF. Comparing the right and left femoral necks through multiple axial CT images showed no obvious differences between the 2 sides (Figures 3, 4). Her only other orthopedic injury was an inferior shoulder dislocation. It is not routine for the general surgery trauma team to obtain a pelvic CT scan prior to involvement of the orthopedic service and prompt reduction of a hip dislocation. Upon initial examination of her right hip, it was fixed in slight flexion and external rotation; she was neurovascularly intact.

After being cleared by the trauma service, the patient provided informed consent for closed reduction of the hip and shoulder under conscious sedation, performed by the emergency department (ED) staff. She received intravenous fentanyl and midazolam, and the reduction was attempted. The reduction maneuver was performed with gentle inline traction, adduction, and internal rotation and extension. There was an audible clunk, and the hip was thought to be reduced and stable. The right leg lower extremity was placed into a knee immobilizer and she remained neurovascularly intact. The shoulder was reduced. After the procedure, the patient had an episode of hypoxia requiring oxygenation via a bag valve mask by the ED staff. Postreduction radiographs confirmed reduction of the right shoulder; however, they also showed a FNF with the femoral head retained near the obturator foramen (Figures 5, 6). The patient and her family were informed of the fracture, and a total hip arthroplasty (THA) was recommended, given her pre-injury mild symptomatic osteoarthritis in the hip and her age. The patient was admitted to the intensive care unit for cardiopulmonary monitoring and was found to have a troponin leak on hospital day 1. She was evaluated by the cardiology service; serial electrocardiograms and troponins ruled out acute myocardial infarction. The patient was cleared for surgery on hospital day 4.

On hospital day 5, she underwent a right THA via a Kocher-Langenbeck approach. The patient’s femoral head was found to be anterior and laterally adjacent to her ischial tuberosity with an indentation fracture. The sciatic nerve was identified and found to be intact. A metal-on-polyethylene Stryker Accolade femoral component and Trident acetabular shell were implanted, and a posterior capsular repair was performed (Figure 7).

The patient tolerated the procedure well, and her postoperative course was uneventful. She was discharged to a subacute rehabilitation facility on postoperative day 3. The patient returned for her 2-week postoperative visit ambulating without assistance. At her last follow-up visit, approximately 6 weeks after surgery, she was a functionally independent community ambulator. Phone conversations with her private orthopedist at 6 months confirmed continued ambulation without problems.

Discussion

This case report of a complication that occurred in our institution has resulted in a change in our protocol for treatment of geriatric anterior hip dislocations. Our institution is a level I trauma center, and traumatic hip dislocations are relatively common, occurring usually in young patients with high-energy trauma. Although somewhat controversial, it is generally assumed that the incidence of avascular necrosis of the femoral head after dislocation of the hip is correlated with the time interval from dislocation to reduction of the hip. Therefore, our protocol for hip dislocations of the hip in young trauma patients is urgent reduction in the ED under appropriate analgesia and muscle relaxation.

In this case report, the patient was older than 65 years with radiographic evidence of possible impingement and postsurgical evidence of impingement of the femoral head in the obturator foremen (Figures 1, 2, 8). In addition, the patient was significantly osteopenic radiographically. An attempted reduction in the ED resulted in FNF requiring THA (Figures 5, 6, 9). After discussion of this complication in our institution’s morbidity and mortality conference, we have developed a protocol for the geriatric patient (older than 65 years) with a traumatic hip dislocation. These patients will undergo attempted reduction under controlled analgesia and muscle relaxation in the operating room (OR) with an attending surgeon present, ideally, an attending surgeon comfortable with arthroplasty in a terminally cleaned OR room. Our institution’s surgical site infection rate after total joint arthroplasty has significantly decreased with improved patient selection and the use of terminally cleaned OR rooms. Because our policy is to perform closed reduction of dislocated hips in an urgent manner, if there is not a terminally clean room or an arthroplasty-trained attending orthopedic surgeon available, then informed consent with discussion of the possibility of fracture requiring a subsequent arthroplasty should be obtained from the patient before the attempted reduction.

After review of the available literature, we believe that this case highlights some of the important treatment principles when treating anterior hip dislocations in the ED. The relatively high incidence of indentation fractures of the femoral head with obturator dislocations puts these fractures at higher risk for possible impingement around the obturator ring. This impingement, coupled with preexisting osteopenia, can predispose these dislocations to FNF, if appropriate analgesia and sedation are not obtained and gentle reduction is not performed. In addition, while it may not be time- or cost-effective to perform closed reduction on every hip dislocation, we bring geriatric patients with radiographic osteopenia to the OR for more controlled reductions. In the informed consent discussion, the possibility of FNF is mentioned, and the patient and family are told that an elective total hip replacement will be performed if this complication occurs.

We consider the following to be risk factors for closed reductions of anterior hip dislocations: (1) preexisting osteopenia on plain films, (2) age greater than 65 years, and (3) radiographic femoral head impingement on the surrounding bony pelvis. We continue to consider closed reduction of both anterior and posterior hip dislocations as urgent (within 6 hours from time of dislocation). This case adds to the existing literature on the risk of FNF with closed reduction of obturator hip dislocations, and we hope that it will encourage further study into the safest and most cost-effective reduction protocol.

Anterior hip dislocations have been reported to account for approximately 5% to 10% of all hip dislocations.¹ Epstein and Wiss² originally divided anterior hip dislocations into superior (type I, including pubic or subspinous) and inferior (type II, including obturator and perineal) dislocations. This classification was further subdivided based on the presence of either no associated fracture (type A), fracture of the femoral head or neck (FNF; type B), or fracture of the acetabulum (type C).³ Of all anterior hip dislocations, it has been reported that the inferior or obturator type of dislocation is more common, constituting approximately 70% of all anterior dislocations.⁴ In 1943, Pringle⁵ described the mechanism of obturator dislocation as simultaneous abduction, flexion, and external rotation of the hip. Our literature search found only 2 case reports in non-English-language journals of a complete FNF associated with an attempted reduction of an anterior hip dislocation.^6,7 Indentation fractures of the femoral head have been more commonly reported than FNFs, with a reported incidence of 35% to 55% after anterior dislocation.^4,8 DeLee and colleagues⁸ also found that those patients with indentation fractures were at a higher risk for developing avascular necrosis of the femoral head in addition to being more likely to report poor or fair function of the hip 2 years after reduction.

There have been a number of different reduction maneuvers for anterior dislocation of hips published in the literature. Epstein and Harvey⁹ advocated reduction by traction in the line of the femur with the hip flexed and in gentle internal rotation and abduction while the patient was under general anesthesia. Toms and Williams,¹⁰ however, recommended adduction with gradual release of the longitudinal traction. Polesky and Polesky¹¹ described a reduction method involving sharp internal rotation, which was found to be associated with FNF. The patient provided written informed consent for print and electronic publication of this case report, and approval was obtained from the Emory University Institutional Review Board.

Case Report

The patient was a 73-year-old woman, an independent ambulator with minimal antecedent hip pain, who, as a pedestrian, was struck by a heavy-duty pickup truck at low velocity. She was flown to our level I trauma center from an outside hospital. The patient arrived hemodynamically stable, with a Glasgow Coma Scale score of 15 and with major complaints of right shoulder and right hip pain. She had a positive Focused Assessment with Sonography for Trauma (FAST), and underwent a subsequent urgent chest, abdomen, and pelvis computed tomography (CT) scan for further investigation. CT showed a grade 1 liver laceration. Her anteroposterior (AP) pelvic radiograph and pelvic CT scan showed an anterior hip dislocation with the femoral head located adjacent to the obturator foramen (Figures 1, 2). The AP pelvic radiograph and pelvic CT scan were scrutinized extensively before reduction to rule out a possible FNF. Comparing the right and left femoral necks through multiple axial CT images showed no obvious differences between the 2 sides (Figures 3, 4). Her only other orthopedic injury was an inferior shoulder dislocation. It is not routine for the general surgery trauma team to obtain a pelvic CT scan prior to involvement of the orthopedic service and prompt reduction of a hip dislocation. Upon initial examination of her right hip, it was fixed in slight flexion and external rotation; she was neurovascularly intact.

After being cleared by the trauma service, the patient provided informed consent for closed reduction of the hip and shoulder under conscious sedation, performed by the emergency department (ED) staff. She received intravenous fentanyl and midazolam, and the reduction was attempted. The reduction maneuver was performed with gentle inline traction, adduction, and internal rotation and extension. There was an audible clunk, and the hip was thought to be reduced and stable. The right leg lower extremity was placed into a knee immobilizer and she remained neurovascularly intact. The shoulder was reduced. After the procedure, the patient had an episode of hypoxia requiring oxygenation via a bag valve mask by the ED staff. Postreduction radiographs confirmed reduction of the right shoulder; however, they also showed a FNF with the femoral head retained near the obturator foramen (Figures 5, 6). The patient and her family were informed of the fracture, and a total hip arthroplasty (THA) was recommended, given her pre-injury mild symptomatic osteoarthritis in the hip and her age. The patient was admitted to the intensive care unit for cardiopulmonary monitoring and was found to have a troponin leak on hospital day 1. She was evaluated by the cardiology service; serial electrocardiograms and troponins ruled out acute myocardial infarction. The patient was cleared for surgery on hospital day 4.

On hospital day 5, she underwent a right THA via a Kocher-Langenbeck approach. The patient’s femoral head was found to be anterior and laterally adjacent to her ischial tuberosity with an indentation fracture. The sciatic nerve was identified and found to be intact. A metal-on-polyethylene Stryker Accolade femoral component and Trident acetabular shell were implanted, and a posterior capsular repair was performed (Figure 7).

The patient tolerated the procedure well, and her postoperative course was uneventful. She was discharged to a subacute rehabilitation facility on postoperative day 3. The patient returned for her 2-week postoperative visit ambulating without assistance. At her last follow-up visit, approximately 6 weeks after surgery, she was a functionally independent community ambulator. Phone conversations with her private orthopedist at 6 months confirmed continued ambulation without problems.

Discussion

This case report of a complication that occurred in our institution has resulted in a change in our protocol for treatment of geriatric anterior hip dislocations. Our institution is a level I trauma center, and traumatic hip dislocations are relatively common, occurring usually in young patients with high-energy trauma. Although somewhat controversial, it is generally assumed that the incidence of avascular necrosis of the femoral head after dislocation of the hip is correlated with the time interval from dislocation to reduction of the hip. Therefore, our protocol for hip dislocations of the hip in young trauma patients is urgent reduction in the ED under appropriate analgesia and muscle relaxation.

In this case report, the patient was older than 65 years with radiographic evidence of possible impingement and postsurgical evidence of impingement of the femoral head in the obturator foremen (Figures 1, 2, 8). In addition, the patient was significantly osteopenic radiographically. An attempted reduction in the ED resulted in FNF requiring THA (Figures 5, 6, 9). After discussion of this complication in our institution’s morbidity and mortality conference, we have developed a protocol for the geriatric patient (older than 65 years) with a traumatic hip dislocation. These patients will undergo attempted reduction under controlled analgesia and muscle relaxation in the operating room (OR) with an attending surgeon present, ideally, an attending surgeon comfortable with arthroplasty in a terminally cleaned OR room. Our institution’s surgical site infection rate after total joint arthroplasty has significantly decreased with improved patient selection and the use of terminally cleaned OR rooms. Because our policy is to perform closed reduction of dislocated hips in an urgent manner, if there is not a terminally clean room or an arthroplasty-trained attending orthopedic surgeon available, then informed consent with discussion of the possibility of fracture requiring a subsequent arthroplasty should be obtained from the patient before the attempted reduction.

After review of the available literature, we believe that this case highlights some of the important treatment principles when treating anterior hip dislocations in the ED. The relatively high incidence of indentation fractures of the femoral head with obturator dislocations puts these fractures at higher risk for possible impingement around the obturator ring. This impingement, coupled with preexisting osteopenia, can predispose these dislocations to FNF, if appropriate analgesia and sedation are not obtained and gentle reduction is not performed. In addition, while it may not be time- or cost-effective to perform closed reduction on every hip dislocation, we bring geriatric patients with radiographic osteopenia to the OR for more controlled reductions. In the informed consent discussion, the possibility of FNF is mentioned, and the patient and family are told that an elective total hip replacement will be performed if this complication occurs.

We consider the following to be risk factors for closed reductions of anterior hip dislocations: (1) preexisting osteopenia on plain films, (2) age greater than 65 years, and (3) radiographic femoral head impingement on the surrounding bony pelvis. We continue to consider closed reduction of both anterior and posterior hip dislocations as urgent (within 6 hours from time of dislocation). This case adds to the existing literature on the risk of FNF with closed reduction of obturator hip dislocations, and we hope that it will encourage further study into the safest and most cost-effective reduction protocol.

Anterior hip dislocations have been reported to account for approximately 5% to 10% of all hip dislocations.¹ Epstein and Wiss² originally divided anterior hip dislocations into superior (type I, including pubic or subspinous) and inferior (type II, including obturator and perineal) dislocations. This classification was further subdivided based on the presence of either no associated fracture (type A), fracture of the femoral head or neck (FNF; type B), or fracture of the acetabulum (type C).³ Of all anterior hip dislocations, it has been reported that the inferior or obturator type of dislocation is more common, constituting approximately 70% of all anterior dislocations.⁴ In 1943, Pringle⁵ described the mechanism of obturator dislocation as simultaneous abduction, flexion, and external rotation of the hip. Our literature search found only 2 case reports in non-English-language journals of a complete FNF associated with an attempted reduction of an anterior hip dislocation.^6,7 Indentation fractures of the femoral head have been more commonly reported than FNFs, with a reported incidence of 35% to 55% after anterior dislocation.^4,8 DeLee and colleagues⁸ also found that those patients with indentation fractures were at a higher risk for developing avascular necrosis of the femoral head in addition to being more likely to report poor or fair function of the hip 2 years after reduction.

There have been a number of different reduction maneuvers for anterior dislocation of hips published in the literature. Epstein and Harvey⁹ advocated reduction by traction in the line of the femur with the hip flexed and in gentle internal rotation and abduction while the patient was under general anesthesia. Toms and Williams,¹⁰ however, recommended adduction with gradual release of the longitudinal traction. Polesky and Polesky¹¹ described a reduction method involving sharp internal rotation, which was found to be associated with FNF. The patient provided written informed consent for print and electronic publication of this case report, and approval was obtained from the Emory University Institutional Review Board.

Case Report

The patient was a 73-year-old woman, an independent ambulator with minimal antecedent hip pain, who, as a pedestrian, was struck by a heavy-duty pickup truck at low velocity. She was flown to our level I trauma center from an outside hospital. The patient arrived hemodynamically stable, with a Glasgow Coma Scale score of 15 and with major complaints of right shoulder and right hip pain. She had a positive Focused Assessment with Sonography for Trauma (FAST), and underwent a subsequent urgent chest, abdomen, and pelvis computed tomography (CT) scan for further investigation. CT showed a grade 1 liver laceration. Her anteroposterior (AP) pelvic radiograph and pelvic CT scan showed an anterior hip dislocation with the femoral head located adjacent to the obturator foramen (Figures 1, 2). The AP pelvic radiograph and pelvic CT scan were scrutinized extensively before reduction to rule out a possible FNF. Comparing the right and left femoral necks through multiple axial CT images showed no obvious differences between the 2 sides (Figures 3, 4). Her only other orthopedic injury was an inferior shoulder dislocation. It is not routine for the general surgery trauma team to obtain a pelvic CT scan prior to involvement of the orthopedic service and prompt reduction of a hip dislocation. Upon initial examination of her right hip, it was fixed in slight flexion and external rotation; she was neurovascularly intact.

After being cleared by the trauma service, the patient provided informed consent for closed reduction of the hip and shoulder under conscious sedation, performed by the emergency department (ED) staff. She received intravenous fentanyl and midazolam, and the reduction was attempted. The reduction maneuver was performed with gentle inline traction, adduction, and internal rotation and extension. There was an audible clunk, and the hip was thought to be reduced and stable. The right leg lower extremity was placed into a knee immobilizer and she remained neurovascularly intact. The shoulder was reduced. After the procedure, the patient had an episode of hypoxia requiring oxygenation via a bag valve mask by the ED staff. Postreduction radiographs confirmed reduction of the right shoulder; however, they also showed a FNF with the femoral head retained near the obturator foramen (Figures 5, 6). The patient and her family were informed of the fracture, and a total hip arthroplasty (THA) was recommended, given her pre-injury mild symptomatic osteoarthritis in the hip and her age. The patient was admitted to the intensive care unit for cardiopulmonary monitoring and was found to have a troponin leak on hospital day 1. She was evaluated by the cardiology service; serial electrocardiograms and troponins ruled out acute myocardial infarction. The patient was cleared for surgery on hospital day 4.

On hospital day 5, she underwent a right THA via a Kocher-Langenbeck approach. The patient’s femoral head was found to be anterior and laterally adjacent to her ischial tuberosity with an indentation fracture. The sciatic nerve was identified and found to be intact. A metal-on-polyethylene Stryker Accolade femoral component and Trident acetabular shell were implanted, and a posterior capsular repair was performed (Figure 7).

The patient tolerated the procedure well, and her postoperative course was uneventful. She was discharged to a subacute rehabilitation facility on postoperative day 3. The patient returned for her 2-week postoperative visit ambulating without assistance. At her last follow-up visit, approximately 6 weeks after surgery, she was a functionally independent community ambulator. Phone conversations with her private orthopedist at 6 months confirmed continued ambulation without problems.

Discussion

This case report of a complication that occurred in our institution has resulted in a change in our protocol for treatment of geriatric anterior hip dislocations. Our institution is a level I trauma center, and traumatic hip dislocations are relatively common, occurring usually in young patients with high-energy trauma. Although somewhat controversial, it is generally assumed that the incidence of avascular necrosis of the femoral head after dislocation of the hip is correlated with the time interval from dislocation to reduction of the hip. Therefore, our protocol for hip dislocations of the hip in young trauma patients is urgent reduction in the ED under appropriate analgesia and muscle relaxation.

In this case report, the patient was older than 65 years with radiographic evidence of possible impingement and postsurgical evidence of impingement of the femoral head in the obturator foremen (Figures 1, 2, 8). In addition, the patient was significantly osteopenic radiographically. An attempted reduction in the ED resulted in FNF requiring THA (Figures 5, 6, 9). After discussion of this complication in our institution’s morbidity and mortality conference, we have developed a protocol for the geriatric patient (older than 65 years) with a traumatic hip dislocation. These patients will undergo attempted reduction under controlled analgesia and muscle relaxation in the operating room (OR) with an attending surgeon present, ideally, an attending surgeon comfortable with arthroplasty in a terminally cleaned OR room. Our institution’s surgical site infection rate after total joint arthroplasty has significantly decreased with improved patient selection and the use of terminally cleaned OR rooms. Because our policy is to perform closed reduction of dislocated hips in an urgent manner, if there is not a terminally clean room or an arthroplasty-trained attending orthopedic surgeon available, then informed consent with discussion of the possibility of fracture requiring a subsequent arthroplasty should be obtained from the patient before the attempted reduction.

After review of the available literature, we believe that this case highlights some of the important treatment principles when treating anterior hip dislocations in the ED. The relatively high incidence of indentation fractures of the femoral head with obturator dislocations puts these fractures at higher risk for possible impingement around the obturator ring. This impingement, coupled with preexisting osteopenia, can predispose these dislocations to FNF, if appropriate analgesia and sedation are not obtained and gentle reduction is not performed. In addition, while it may not be time- or cost-effective to perform closed reduction on every hip dislocation, we bring geriatric patients with radiographic osteopenia to the OR for more controlled reductions. In the informed consent discussion, the possibility of FNF is mentioned, and the patient and family are told that an elective total hip replacement will be performed if this complication occurs.

We consider the following to be risk factors for closed reductions of anterior hip dislocations: (1) preexisting osteopenia on plain films, (2) age greater than 65 years, and (3) radiographic femoral head impingement on the surrounding bony pelvis. We continue to consider closed reduction of both anterior and posterior hip dislocations as urgent (within 6 hours from time of dislocation). This case adds to the existing literature on the risk of FNF with closed reduction of obturator hip dislocations, and we hope that it will encourage further study into the safest and most cost-effective reduction protocol.

The biceps brachii derives its name from the 2 heads of the muscle. The short head originates from the coracoid apex, with the coracobrachialis muscle. The long head of the biceps tendon (LHBT) starts within the capsule of the shoulder joint, running from the supraglenoid tubercle or labrum.¹ The tendon typically runs free along its intra-articular course, but it is also extrasynovial and ensheathed by a continuation of the synovial lining of the articular capsule that extends to the inferior-most extent of the bicipital groove.² Congenital anomalies of the LHBT are uncommon, although several atypical forms have been described. A literature search for anomalous LHBT identified several variations in anatomic descriptions, including Y-shaped variant, complete absence of tendon, extra-articular attachment, and a variety of intracapsular attachments. In all, 8 case reports of aberrant intracapsular attachment of LHBT^3-12 were identified. These cases presented with a variety of clinical manifestations and pathologic changes. Often, these anatomic variations are considered innocuous, yet some present with pathologic findings.

We present the clinical, magnetic resonance imaging (MRI), and arthroscopic findings of a relatively young athletic patient who was experiencing symptoms of bilateral superior labrum anterior to posterior (SLAP) tears that were unresponsive to conservative management. A unique anatomic variant of the LHBT that involved confluence of the LHBT with the undersurface of the anterosuperior capsule at the rotator interval, as well as a Buford complex anteriorly, was identified and treated. We believe that the tethering of the biceps tendon to the capsule combined with the Buford complex created increased stress on the superior labrum and biceps anchor variant, leading to the development of bilateral symptomatic type II SLAP tears. Knowledge of this variant, though perhaps rare, may be relevant for diagnostic recognition of young athletic patients who present with recalcitrant shoulder symptoms. The patient and the patient’s parents provided written informed consent for print and electronic publication of this case report.

Case Report

A 15-year-old healthy and active athletic boy presented with pain in the right shoulder without history of trauma. He was active in both swimming and baseball. He complained of pain that was present with activities, such as lifting weights, swimming, and throwing. His treatment prior to the office visit consisted of nonsteroidal anti-inflammatory medication, rest, and a therapy program initiated by his high school athletic trainer.

Physical examination demonstrated tenderness to palpation over the posterior capsule and biceps. Motion was full, cuff strength was normal, and SLAP signs (O’Brien, Speed, and Jobe relocation) were positive. A radiograph showed no sign of fracture or dislocation, and no evidence of bony abnormality.

The patient was sent for an MRI arthrogram, which showed a SLAP tear extending from 1 o’clock anteriorly to 10 o’clock posteriorly without intra-articular displacement. No rotator cuff tear was noted. The biceps tendon was noted to be unremarkable and located within the bicipital groove, although retrospective review of the MRI showed that the intra-articular biceps tendon was somewhat confluent with the adjacent tissues.

The patient underwent right shoulder arthroscopy. The shoulder was stable to ligamentous examination under anesthesia. Arthroscopic evaluation revealed that there was a type II SLAP tear extending from the 11-o’clock to the 2-o’clock positions. The superior glenohumeral ligament was identified as it arose from the upper pole of the glenoid labrum and then ran parallel and inferior to the tendon of the biceps towards the lesser tubercle. Surprisingly, there was a very unusual attachment of the intracapsular LHBT to the undersurface of the rotator interval, which restricted biceps excursion in relation to the rotator cuff. Additionally, there was a thick cord-like middle glenohumeral ligament anteriorly that lacked the normal glenoid attachments, thus representing a Buford complex. Interestingly, the labral tear could not only be displaced with a probe, but placing the shoulder through a range of motion also led to increased displacement of the labrum from the glenoid, likely because the biceps tendon was tethered to the undersurface of the capsule.

At the time of arthroscopy, the LHBT was released from its attachment to the capsule at the rotator interval with a radiofrequency wand and shaver. A labral repair was performed using three 2.9-mm bioabsorbable suture anchors, placing 2 posterior and 1 anterior to the biceps tendon. The integrity of the labral repair was observed while placing the shoulder through range of motion.  

Postoperatively, the patient was kept in a sling for 5 weeks. Home exercises were initiated at 2 weeks, and outpatient physical therapy was implemented at 4 weeks. The patient resumed swimming, throwing, and other activities—with minimal discomfort—at 6 months postoperatively.

Three years after his initial visit, the patient returned to the office with a similar complaint of pain and limitation of function in his left shoulder after returning to full athletic competition. Once again, there was no history of injury, and history, physical examination, and MRI arthrogram (Figures 1A, 1B) evaluation proved to be very similar to this young athlete’s right shoulder work-up.

The patient once again underwent shoulder arthroscopy and treatment. Although this was now the left shoulder, the findings were essentially identical to the right shoulder. Once again, the labrum was detached from the 11-o’clock to 2-o’clock positions, and a Buford complex was present anteriorly (Figure 2A). The labral tear was easily displaceable from the glenoid with a probe, and placing the shoulder through a range of motion led to increased displacement of the labrum from the glenoid. There was also confluence of the intra-articular LHBT with the undersurface of the capsule within the rotator interval (Figure 2B). A radiofrequency wand, shaver, and elevator were used to define the biceps tendon and separate it from the undersurface of the capsule. The SLAP repair was performed using three 2.9-mm absorbable suture anchors with 2 posterior and 1 anterior to the biceps tendon insertion. The labral repair was observed while placing the shoulder through range of motion and the shoulder was seen to be free of any undue tension on the labrum.

Postoperatively, the patient’s sling and rehabilitation protocol was identical to that of the right shoulder. The patient progressed well, was released to full activity at 6 months, and has not returned with any further complaints of left or right shoulder pain. Approximately 3 years after treatment the patient was contacted via phone and asked about symptoms, pain, and activity. He denies current symptoms of clicking or instability and has no pain that he can identify as being related to previous pathology or treatment. Since the surgery, he has ceased competitive sports and weight lifting, which he attributes to deconditioning associated with postsurgical immobilization and lack of motivation.

Discussion

Of the 8 case reports in the literature that identified variable intra-articular biceps insertional anatomy, only 2 reports represented confluence of the biceps within the rotator interval.⁷ Interestingly, of the cases identified, the single case that presented a patient with similar pathology of a type II SLAP lesion had an almost identical anatomical variant presentation consisting of both the anomalous insertion of the LHBT into the undersurface of the rotator interval and a Buford variant of the anterosuperior glenohumeral ligament complex. To our knowledge, our bilateral case of an altered intra-articular biceps insertion and a concomitant SLAP tear supports the theory that this pattern of anomalous insertion may very well have altered the biomechanics of the tendon, resulting in acquired pathology to the superior labrum.

The literature reviewed showed the prevalence of anatomic variations of the LHBT ranged from 1.9% to 7.4%.^13,14 These variations are generally considered benign; however, in some cases—as in the cases of the young athletes presented by Wahl and MacGillivray⁷ and in this report—anatomic variation may play an important role in pathogenesis of different injury patterns. The primary function of the LHBT is the stabilization of the glenohumeral joint during abduction and external rotation.¹⁵ When the insertion diverges from normal (eg, when the tendon is tethered to the undersurface of the rotator cuff), the biomechanical stresses on the tendon likely change. As a result of the anomalous position of the LHBT origin, there may be a change in the shoulder joint’s biomechanics, with increased strain on the glenohumeral ligament and its attachment onto the glenoid.¹⁶

This case report differs from publications on variable superior glenohumeral ligament attachments because a discrete superior glenohumeral ligament structure was isolated from the biceps tendon. Although a larger case series or patient cohort, as well as more involved biomechanical analysis, would certainly be necessary to prove our hypothesis, we believe that this case suggests certain anatomic LHBT and labral variations can contribute to the develop of SLAP tears in younger individuals.

The biceps brachii derives its name from the 2 heads of the muscle. The short head originates from the coracoid apex, with the coracobrachialis muscle. The long head of the biceps tendon (LHBT) starts within the capsule of the shoulder joint, running from the supraglenoid tubercle or labrum.¹ The tendon typically runs free along its intra-articular course, but it is also extrasynovial and ensheathed by a continuation of the synovial lining of the articular capsule that extends to the inferior-most extent of the bicipital groove.² Congenital anomalies of the LHBT are uncommon, although several atypical forms have been described. A literature search for anomalous LHBT identified several variations in anatomic descriptions, including Y-shaped variant, complete absence of tendon, extra-articular attachment, and a variety of intracapsular attachments. In all, 8 case reports of aberrant intracapsular attachment of LHBT^3-12 were identified. These cases presented with a variety of clinical manifestations and pathologic changes. Often, these anatomic variations are considered innocuous, yet some present with pathologic findings.

We present the clinical, magnetic resonance imaging (MRI), and arthroscopic findings of a relatively young athletic patient who was experiencing symptoms of bilateral superior labrum anterior to posterior (SLAP) tears that were unresponsive to conservative management. A unique anatomic variant of the LHBT that involved confluence of the LHBT with the undersurface of the anterosuperior capsule at the rotator interval, as well as a Buford complex anteriorly, was identified and treated. We believe that the tethering of the biceps tendon to the capsule combined with the Buford complex created increased stress on the superior labrum and biceps anchor variant, leading to the development of bilateral symptomatic type II SLAP tears. Knowledge of this variant, though perhaps rare, may be relevant for diagnostic recognition of young athletic patients who present with recalcitrant shoulder symptoms. The patient and the patient’s parents provided written informed consent for print and electronic publication of this case report.

Case Report

A 15-year-old healthy and active athletic boy presented with pain in the right shoulder without history of trauma. He was active in both swimming and baseball. He complained of pain that was present with activities, such as lifting weights, swimming, and throwing. His treatment prior to the office visit consisted of nonsteroidal anti-inflammatory medication, rest, and a therapy program initiated by his high school athletic trainer.

Physical examination demonstrated tenderness to palpation over the posterior capsule and biceps. Motion was full, cuff strength was normal, and SLAP signs (O’Brien, Speed, and Jobe relocation) were positive. A radiograph showed no sign of fracture or dislocation, and no evidence of bony abnormality.

The patient was sent for an MRI arthrogram, which showed a SLAP tear extending from 1 o’clock anteriorly to 10 o’clock posteriorly without intra-articular displacement. No rotator cuff tear was noted. The biceps tendon was noted to be unremarkable and located within the bicipital groove, although retrospective review of the MRI showed that the intra-articular biceps tendon was somewhat confluent with the adjacent tissues.

The patient underwent right shoulder arthroscopy. The shoulder was stable to ligamentous examination under anesthesia. Arthroscopic evaluation revealed that there was a type II SLAP tear extending from the 11-o’clock to the 2-o’clock positions. The superior glenohumeral ligament was identified as it arose from the upper pole of the glenoid labrum and then ran parallel and inferior to the tendon of the biceps towards the lesser tubercle. Surprisingly, there was a very unusual attachment of the intracapsular LHBT to the undersurface of the rotator interval, which restricted biceps excursion in relation to the rotator cuff. Additionally, there was a thick cord-like middle glenohumeral ligament anteriorly that lacked the normal glenoid attachments, thus representing a Buford complex. Interestingly, the labral tear could not only be displaced with a probe, but placing the shoulder through a range of motion also led to increased displacement of the labrum from the glenoid, likely because the biceps tendon was tethered to the undersurface of the capsule.

At the time of arthroscopy, the LHBT was released from its attachment to the capsule at the rotator interval with a radiofrequency wand and shaver. A labral repair was performed using three 2.9-mm bioabsorbable suture anchors, placing 2 posterior and 1 anterior to the biceps tendon. The integrity of the labral repair was observed while placing the shoulder through range of motion.  

Postoperatively, the patient was kept in a sling for 5 weeks. Home exercises were initiated at 2 weeks, and outpatient physical therapy was implemented at 4 weeks. The patient resumed swimming, throwing, and other activities—with minimal discomfort—at 6 months postoperatively.

Three years after his initial visit, the patient returned to the office with a similar complaint of pain and limitation of function in his left shoulder after returning to full athletic competition. Once again, there was no history of injury, and history, physical examination, and MRI arthrogram (Figures 1A, 1B) evaluation proved to be very similar to this young athlete’s right shoulder work-up.

The patient once again underwent shoulder arthroscopy and treatment. Although this was now the left shoulder, the findings were essentially identical to the right shoulder. Once again, the labrum was detached from the 11-o’clock to 2-o’clock positions, and a Buford complex was present anteriorly (Figure 2A). The labral tear was easily displaceable from the glenoid with a probe, and placing the shoulder through a range of motion led to increased displacement of the labrum from the glenoid. There was also confluence of the intra-articular LHBT with the undersurface of the capsule within the rotator interval (Figure 2B). A radiofrequency wand, shaver, and elevator were used to define the biceps tendon and separate it from the undersurface of the capsule. The SLAP repair was performed using three 2.9-mm absorbable suture anchors with 2 posterior and 1 anterior to the biceps tendon insertion. The labral repair was observed while placing the shoulder through range of motion and the shoulder was seen to be free of any undue tension on the labrum.

Postoperatively, the patient’s sling and rehabilitation protocol was identical to that of the right shoulder. The patient progressed well, was released to full activity at 6 months, and has not returned with any further complaints of left or right shoulder pain. Approximately 3 years after treatment the patient was contacted via phone and asked about symptoms, pain, and activity. He denies current symptoms of clicking or instability and has no pain that he can identify as being related to previous pathology or treatment. Since the surgery, he has ceased competitive sports and weight lifting, which he attributes to deconditioning associated with postsurgical immobilization and lack of motivation.

Discussion

Of the 8 case reports in the literature that identified variable intra-articular biceps insertional anatomy, only 2 reports represented confluence of the biceps within the rotator interval.⁷ Interestingly, of the cases identified, the single case that presented a patient with similar pathology of a type II SLAP lesion had an almost identical anatomical variant presentation consisting of both the anomalous insertion of the LHBT into the undersurface of the rotator interval and a Buford variant of the anterosuperior glenohumeral ligament complex. To our knowledge, our bilateral case of an altered intra-articular biceps insertion and a concomitant SLAP tear supports the theory that this pattern of anomalous insertion may very well have altered the biomechanics of the tendon, resulting in acquired pathology to the superior labrum.

The literature reviewed showed the prevalence of anatomic variations of the LHBT ranged from 1.9% to 7.4%.^13,14 These variations are generally considered benign; however, in some cases—as in the cases of the young athletes presented by Wahl and MacGillivray⁷ and in this report—anatomic variation may play an important role in pathogenesis of different injury patterns. The primary function of the LHBT is the stabilization of the glenohumeral joint during abduction and external rotation.¹⁵ When the insertion diverges from normal (eg, when the tendon is tethered to the undersurface of the rotator cuff), the biomechanical stresses on the tendon likely change. As a result of the anomalous position of the LHBT origin, there may be a change in the shoulder joint’s biomechanics, with increased strain on the glenohumeral ligament and its attachment onto the glenoid.¹⁶

This case report differs from publications on variable superior glenohumeral ligament attachments because a discrete superior glenohumeral ligament structure was isolated from the biceps tendon. Although a larger case series or patient cohort, as well as more involved biomechanical analysis, would certainly be necessary to prove our hypothesis, we believe that this case suggests certain anatomic LHBT and labral variations can contribute to the develop of SLAP tears in younger individuals.

The biceps brachii derives its name from the 2 heads of the muscle. The short head originates from the coracoid apex, with the coracobrachialis muscle. The long head of the biceps tendon (LHBT) starts within the capsule of the shoulder joint, running from the supraglenoid tubercle or labrum.¹ The tendon typically runs free along its intra-articular course, but it is also extrasynovial and ensheathed by a continuation of the synovial lining of the articular capsule that extends to the inferior-most extent of the bicipital groove.² Congenital anomalies of the LHBT are uncommon, although several atypical forms have been described. A literature search for anomalous LHBT identified several variations in anatomic descriptions, including Y-shaped variant, complete absence of tendon, extra-articular attachment, and a variety of intracapsular attachments. In all, 8 case reports of aberrant intracapsular attachment of LHBT^3-12 were identified. These cases presented with a variety of clinical manifestations and pathologic changes. Often, these anatomic variations are considered innocuous, yet some present with pathologic findings.

We present the clinical, magnetic resonance imaging (MRI), and arthroscopic findings of a relatively young athletic patient who was experiencing symptoms of bilateral superior labrum anterior to posterior (SLAP) tears that were unresponsive to conservative management. A unique anatomic variant of the LHBT that involved confluence of the LHBT with the undersurface of the anterosuperior capsule at the rotator interval, as well as a Buford complex anteriorly, was identified and treated. We believe that the tethering of the biceps tendon to the capsule combined with the Buford complex created increased stress on the superior labrum and biceps anchor variant, leading to the development of bilateral symptomatic type II SLAP tears. Knowledge of this variant, though perhaps rare, may be relevant for diagnostic recognition of young athletic patients who present with recalcitrant shoulder symptoms. The patient and the patient’s parents provided written informed consent for print and electronic publication of this case report.

Case Report

A 15-year-old healthy and active athletic boy presented with pain in the right shoulder without history of trauma. He was active in both swimming and baseball. He complained of pain that was present with activities, such as lifting weights, swimming, and throwing. His treatment prior to the office visit consisted of nonsteroidal anti-inflammatory medication, rest, and a therapy program initiated by his high school athletic trainer.

Physical examination demonstrated tenderness to palpation over the posterior capsule and biceps. Motion was full, cuff strength was normal, and SLAP signs (O’Brien, Speed, and Jobe relocation) were positive. A radiograph showed no sign of fracture or dislocation, and no evidence of bony abnormality.

The patient was sent for an MRI arthrogram, which showed a SLAP tear extending from 1 o’clock anteriorly to 10 o’clock posteriorly without intra-articular displacement. No rotator cuff tear was noted. The biceps tendon was noted to be unremarkable and located within the bicipital groove, although retrospective review of the MRI showed that the intra-articular biceps tendon was somewhat confluent with the adjacent tissues.

The patient underwent right shoulder arthroscopy. The shoulder was stable to ligamentous examination under anesthesia. Arthroscopic evaluation revealed that there was a type II SLAP tear extending from the 11-o’clock to the 2-o’clock positions. The superior glenohumeral ligament was identified as it arose from the upper pole of the glenoid labrum and then ran parallel and inferior to the tendon of the biceps towards the lesser tubercle. Surprisingly, there was a very unusual attachment of the intracapsular LHBT to the undersurface of the rotator interval, which restricted biceps excursion in relation to the rotator cuff. Additionally, there was a thick cord-like middle glenohumeral ligament anteriorly that lacked the normal glenoid attachments, thus representing a Buford complex. Interestingly, the labral tear could not only be displaced with a probe, but placing the shoulder through a range of motion also led to increased displacement of the labrum from the glenoid, likely because the biceps tendon was tethered to the undersurface of the capsule.

At the time of arthroscopy, the LHBT was released from its attachment to the capsule at the rotator interval with a radiofrequency wand and shaver. A labral repair was performed using three 2.9-mm bioabsorbable suture anchors, placing 2 posterior and 1 anterior to the biceps tendon. The integrity of the labral repair was observed while placing the shoulder through range of motion.  

Postoperatively, the patient was kept in a sling for 5 weeks. Home exercises were initiated at 2 weeks, and outpatient physical therapy was implemented at 4 weeks. The patient resumed swimming, throwing, and other activities—with minimal discomfort—at 6 months postoperatively.

Three years after his initial visit, the patient returned to the office with a similar complaint of pain and limitation of function in his left shoulder after returning to full athletic competition. Once again, there was no history of injury, and history, physical examination, and MRI arthrogram (Figures 1A, 1B) evaluation proved to be very similar to this young athlete’s right shoulder work-up.

The patient once again underwent shoulder arthroscopy and treatment. Although this was now the left shoulder, the findings were essentially identical to the right shoulder. Once again, the labrum was detached from the 11-o’clock to 2-o’clock positions, and a Buford complex was present anteriorly (Figure 2A). The labral tear was easily displaceable from the glenoid with a probe, and placing the shoulder through a range of motion led to increased displacement of the labrum from the glenoid. There was also confluence of the intra-articular LHBT with the undersurface of the capsule within the rotator interval (Figure 2B). A radiofrequency wand, shaver, and elevator were used to define the biceps tendon and separate it from the undersurface of the capsule. The SLAP repair was performed using three 2.9-mm absorbable suture anchors with 2 posterior and 1 anterior to the biceps tendon insertion. The labral repair was observed while placing the shoulder through range of motion and the shoulder was seen to be free of any undue tension on the labrum.

Postoperatively, the patient’s sling and rehabilitation protocol was identical to that of the right shoulder. The patient progressed well, was released to full activity at 6 months, and has not returned with any further complaints of left or right shoulder pain. Approximately 3 years after treatment the patient was contacted via phone and asked about symptoms, pain, and activity. He denies current symptoms of clicking or instability and has no pain that he can identify as being related to previous pathology or treatment. Since the surgery, he has ceased competitive sports and weight lifting, which he attributes to deconditioning associated with postsurgical immobilization and lack of motivation.

Discussion

Of the 8 case reports in the literature that identified variable intra-articular biceps insertional anatomy, only 2 reports represented confluence of the biceps within the rotator interval.⁷ Interestingly, of the cases identified, the single case that presented a patient with similar pathology of a type II SLAP lesion had an almost identical anatomical variant presentation consisting of both the anomalous insertion of the LHBT into the undersurface of the rotator interval and a Buford variant of the anterosuperior glenohumeral ligament complex. To our knowledge, our bilateral case of an altered intra-articular biceps insertion and a concomitant SLAP tear supports the theory that this pattern of anomalous insertion may very well have altered the biomechanics of the tendon, resulting in acquired pathology to the superior labrum.

The literature reviewed showed the prevalence of anatomic variations of the LHBT ranged from 1.9% to 7.4%.^13,14 These variations are generally considered benign; however, in some cases—as in the cases of the young athletes presented by Wahl and MacGillivray⁷ and in this report—anatomic variation may play an important role in pathogenesis of different injury patterns. The primary function of the LHBT is the stabilization of the glenohumeral joint during abduction and external rotation.¹⁵ When the insertion diverges from normal (eg, when the tendon is tethered to the undersurface of the rotator cuff), the biomechanical stresses on the tendon likely change. As a result of the anomalous position of the LHBT origin, there may be a change in the shoulder joint’s biomechanics, with increased strain on the glenohumeral ligament and its attachment onto the glenoid.¹⁶

This case report differs from publications on variable superior glenohumeral ligament attachments because a discrete superior glenohumeral ligament structure was isolated from the biceps tendon. Although a larger case series or patient cohort, as well as more involved biomechanical analysis, would certainly be necessary to prove our hypothesis, we believe that this case suggests certain anatomic LHBT and labral variations can contribute to the develop of SLAP tears in younger individuals.

Xanthogranulomatous osteomyelitis (XO) is a type of chronic inflammatory process that is characterized by the collection of foamy macrophages along with mononuclear cells in the tissue.¹ Xanthogranulomatous osteomyelitis is characterized by the presence of granular, eosinophilic, periodic acid–Schiff–positive histiocytes in the initial stages, followed by the mixture of foamy macrophages and activated plasma cells and, last, by the presence of suppurative foci and hemorrhage. This is an uncommon process best known to occur in the gallbladder, kidney, urinary bladder, fallopian tube, ovary, vagina, prostate, testis, epididymis, colon, and appendix.^2-4 Very rarely, it can affect lungs, brain, or bone. Only 5 cases of XO have been reported in the literature.^5-8

We report XO of the proximal femur in a 65-year-old woman who initially had a clinical and radiologic diagnosis of aneurysmal bone cyst; however, histopathologic examination confirmed the diagnosis of XO. Xanthogranulomatous osteomyelitis mimics a neoplastic pathology in gallbladder, kidney, and prostrate on gross clinical and radiologic examination.⁹ The pathogenesis of XO is best characterized by a delayed type of hypersensitivity reaction.¹⁰ The differential diagnosis includes chronic recurrent multifocal osteomyelitis, xanthoma, infiltrative storage disorder, malakoplakia, Langerhans cell histiocytosis, fibrohistiocytic tumor, Erdheim-Chester disease, and metastatic renal cell carcinoma.^11-14 The patient provided written informed consent for print and electronic publication of this case report.

Case Report

A 65-year-old hypertensive woman presented with complaints of pain in the right hip for a duration of 6 months. Pain was radiating from the right hip region to the anteromedial aspect of the knee and progressively increasing, with a history of pain at rest suggestive of a nonmechanical pathology in the hip. There was no history of fever, weight loss, loss of appetite, pain in any other joint, or morning stiffness. The patient was mobile without support and was able to squat and sit cross-legged; however, the stance phase on the right side was less than on the left side, suggestive of an antalgic component in the gait.

On examining the patient, there was anterior hip joint tenderness with no local sign of any infective or inflammatory pathology. Trochanteric tenderness was present, but there was no irregularity, broadening, or thickening of the trochanter. There was no restriction in the range of motion, and no coronal or sagittal plane deformity in the right hip. There was no limb-length discrepancy. However, the patient was not able to raise her leg actively, probably because of pain in the right hip.

On plain radiographs of the pelvis with bilateral hips, a well-defined nonexpansile uniloculated lytic lesion with sclerotic margins was present in the neck of the right femur, extending to the intertrochanteric area (Figure 1). Ground-glass appearance was also noted. Considering the benign nature of the lesion radiologically and clinically, a differential diagnosis of hyperparathyroidism, renal osteodystrophy, multiple myeloma, and fibrous dysplasia was considered. Hematologic investigations, skeletal survey, and magnetic resonance imaging (MRI) of the bilateral hips were performed to rule out the differential diagnosis.

The patient’s hemoglobin level was 11.8 g/dL with total white blood cell count of 10,300/µL. Renal and hepatic functions were within normal limit. Serum erythrocyte sedimentation rate (ESR) was 12 mm/h and C-reactive protein level was normal. Serum parathyroid level was 32 pg/mL, which was within normal limits, with an alkaline phosphatase level of 101 U/L. The skeletal survey showed no other bony lesion in the body. T1-weighted MRI of both hips showed a well-defined hypointense lesion in the neck and intertrochanteric area of the right hip, which was hyperintense on T2-weighted MRI, suggestive of aneurysmal bone cyst (Figure 2).

Normal ESR, hemoglobin, alkaline phosphatase, and serum parathyroid levels and normal skeletal survey almost ruled out multiple myeloma and hyperparathyroidism. Normal renal profile ruled out renal osteodystrophy and the osteitis fibrosa cystica lesion associated with it. We planned for prophylactic internal fixation of the lesion to prevent a pathologic fracture. According to Mirels,¹⁵ if there is a lytic lesion covering more than two-thirds of the circumference of the bone in the peritrochanteric area, the chances of a pathologic fracture are high and such fractures should be fixed.

We planned for curettage of the lesion with bone grafting and in situ intramedullary fixation of the lesion. Curettage was done according to the plan and the sample was sent for histopathologic examination. In situ internal fixation and bone grafting were performed by using a proximal femoral intramedullary nail. To our surprise, the biopsy sample was reported as xanthogranuloma, with multiple foamy macrophages mixed with inflammatory cells and aggregates of lymphocytes (Figure 3). Mycobacterial and routine bacterial cultures were reported as negative. The patient was kept on oral antibiotics (cefixime and moxifloxacin) for 6 weeks, and she made an uneventful recovery. At 6-month follow-up, a radiograph of the right hip showed a healed lesion with proximal femoral nail in situ (Figure 4).

Discussion

To the best of our knowledge, a total of 5 cases of XO have been reported in the literature. The earliest of these reports were by Cozzutto and Carbone,¹ who reported 2 cases of XO of the first rib and of the epiphysis of the tibia, respectively. The importance of these lesions to diagnosis is their confusion with a neoplastic disease, as XO is itself a benign disorder. These lesions can mimic a neoplastic lesion in clinical and radiologic presentation and the only way to differentiate the lesion from a neoplastic disease is by histopathologic examination of the tissue. Hypothetically, xanthogranulomatous disorders can be related to trauma or infection.

In 2007, Vankalakunti and colleagues⁶ reported XO of the ulna in a 50-year-old postmenopausal woman. In that case, progressive swelling was present on the extensor aspect of her right forearm for a period of 2 years, for which curettage and bone grafting were performed, using autograft from the ipsilateral iliac crest. The tissue culture was sterile, and XO was diagnosed as a result of the histopathologic examination. In 2009, Cennimo and colleagues⁷ reported XO of the index finger and wrist of a man complaining of pain and swelling for 1 year, which was unresponsive to antibiotics. The diagnosis of XO was confirmed histopathologically, when the culture of the same tissue grew Mycobacterium marinum. Radical synovectomy of the lesion was performed, after which minocycline, clarithromycin, and ethambutol were administered. In 2012, Borjian and colleagues⁸ reported a case of XO of the proximal humerus and proximal fibula in a 14-year-old child. The child, who presented with fever, pain, and restriction of shoulder movements, was started on oral antibiotics as the tissue culture grew Staphylococcus aureus; the patient did not complete the course of treatment in the hospital. No surgical intervention was done in this case. The diagnosis of XO was confirmed by microscopic examination of the tissue.

An association between bacterial infection and xanthogranulomatous inflammation has existed in several organs, such as the kidneys, and in the gastrointestinal system, but such an association of the 2 is yet to be determined for bone.^5,10,16-19 Because of the paucity of literature on the disease, a management protocol for XO of bone has not been defined, and decisions have to be made considering the natural history of the disease in other organs. We present this case primarily because of its rarity, curability, and its close resemblance to bone tumors. While XO is benign, it can mimic a neoplastic bone lesion in its imaging and clinical manifestations, and appropriate differentiation is crucial. Currently, histopathologic examination of lesions is the most specific and is the gold standard for diagnosis.

Conclusion

Xanthogranulomatous osteomyelitis is a very rare entity, and only a few cases have been reported in the English-language literature. Though rare, XO warrants greater emphasis than it receives in the literature. It is a chronic inflammatory disease having a close resemblance to bone tumors. A high index of suspicion must be practiced to differentiate XO from tumors. Histopathologic examination is mandatory to establish definitive diagnosis and correct treatment.

Xanthogranulomatous osteomyelitis (XO) is a type of chronic inflammatory process that is characterized by the collection of foamy macrophages along with mononuclear cells in the tissue.¹ Xanthogranulomatous osteomyelitis is characterized by the presence of granular, eosinophilic, periodic acid–Schiff–positive histiocytes in the initial stages, followed by the mixture of foamy macrophages and activated plasma cells and, last, by the presence of suppurative foci and hemorrhage. This is an uncommon process best known to occur in the gallbladder, kidney, urinary bladder, fallopian tube, ovary, vagina, prostate, testis, epididymis, colon, and appendix.^2-4 Very rarely, it can affect lungs, brain, or bone. Only 5 cases of XO have been reported in the literature.^5-8

We report XO of the proximal femur in a 65-year-old woman who initially had a clinical and radiologic diagnosis of aneurysmal bone cyst; however, histopathologic examination confirmed the diagnosis of XO. Xanthogranulomatous osteomyelitis mimics a neoplastic pathology in gallbladder, kidney, and prostrate on gross clinical and radiologic examination.⁹ The pathogenesis of XO is best characterized by a delayed type of hypersensitivity reaction.¹⁰ The differential diagnosis includes chronic recurrent multifocal osteomyelitis, xanthoma, infiltrative storage disorder, malakoplakia, Langerhans cell histiocytosis, fibrohistiocytic tumor, Erdheim-Chester disease, and metastatic renal cell carcinoma.^11-14 The patient provided written informed consent for print and electronic publication of this case report.

Case Report

A 65-year-old hypertensive woman presented with complaints of pain in the right hip for a duration of 6 months. Pain was radiating from the right hip region to the anteromedial aspect of the knee and progressively increasing, with a history of pain at rest suggestive of a nonmechanical pathology in the hip. There was no history of fever, weight loss, loss of appetite, pain in any other joint, or morning stiffness. The patient was mobile without support and was able to squat and sit cross-legged; however, the stance phase on the right side was less than on the left side, suggestive of an antalgic component in the gait.

On examining the patient, there was anterior hip joint tenderness with no local sign of any infective or inflammatory pathology. Trochanteric tenderness was present, but there was no irregularity, broadening, or thickening of the trochanter. There was no restriction in the range of motion, and no coronal or sagittal plane deformity in the right hip. There was no limb-length discrepancy. However, the patient was not able to raise her leg actively, probably because of pain in the right hip.

On plain radiographs of the pelvis with bilateral hips, a well-defined nonexpansile uniloculated lytic lesion with sclerotic margins was present in the neck of the right femur, extending to the intertrochanteric area (Figure 1). Ground-glass appearance was also noted. Considering the benign nature of the lesion radiologically and clinically, a differential diagnosis of hyperparathyroidism, renal osteodystrophy, multiple myeloma, and fibrous dysplasia was considered. Hematologic investigations, skeletal survey, and magnetic resonance imaging (MRI) of the bilateral hips were performed to rule out the differential diagnosis.

The patient’s hemoglobin level was 11.8 g/dL with total white blood cell count of 10,300/µL. Renal and hepatic functions were within normal limit. Serum erythrocyte sedimentation rate (ESR) was 12 mm/h and C-reactive protein level was normal. Serum parathyroid level was 32 pg/mL, which was within normal limits, with an alkaline phosphatase level of 101 U/L. The skeletal survey showed no other bony lesion in the body. T1-weighted MRI of both hips showed a well-defined hypointense lesion in the neck and intertrochanteric area of the right hip, which was hyperintense on T2-weighted MRI, suggestive of aneurysmal bone cyst (Figure 2).

Normal ESR, hemoglobin, alkaline phosphatase, and serum parathyroid levels and normal skeletal survey almost ruled out multiple myeloma and hyperparathyroidism. Normal renal profile ruled out renal osteodystrophy and the osteitis fibrosa cystica lesion associated with it. We planned for prophylactic internal fixation of the lesion to prevent a pathologic fracture. According to Mirels,¹⁵ if there is a lytic lesion covering more than two-thirds of the circumference of the bone in the peritrochanteric area, the chances of a pathologic fracture are high and such fractures should be fixed.

We planned for curettage of the lesion with bone grafting and in situ intramedullary fixation of the lesion. Curettage was done according to the plan and the sample was sent for histopathologic examination. In situ internal fixation and bone grafting were performed by using a proximal femoral intramedullary nail. To our surprise, the biopsy sample was reported as xanthogranuloma, with multiple foamy macrophages mixed with inflammatory cells and aggregates of lymphocytes (Figure 3). Mycobacterial and routine bacterial cultures were reported as negative. The patient was kept on oral antibiotics (cefixime and moxifloxacin) for 6 weeks, and she made an uneventful recovery. At 6-month follow-up, a radiograph of the right hip showed a healed lesion with proximal femoral nail in situ (Figure 4).

Discussion

To the best of our knowledge, a total of 5 cases of XO have been reported in the literature. The earliest of these reports were by Cozzutto and Carbone,¹ who reported 2 cases of XO of the first rib and of the epiphysis of the tibia, respectively. The importance of these lesions to diagnosis is their confusion with a neoplastic disease, as XO is itself a benign disorder. These lesions can mimic a neoplastic lesion in clinical and radiologic presentation and the only way to differentiate the lesion from a neoplastic disease is by histopathologic examination of the tissue. Hypothetically, xanthogranulomatous disorders can be related to trauma or infection.

In 2007, Vankalakunti and colleagues⁶ reported XO of the ulna in a 50-year-old postmenopausal woman. In that case, progressive swelling was present on the extensor aspect of her right forearm for a period of 2 years, for which curettage and bone grafting were performed, using autograft from the ipsilateral iliac crest. The tissue culture was sterile, and XO was diagnosed as a result of the histopathologic examination. In 2009, Cennimo and colleagues⁷ reported XO of the index finger and wrist of a man complaining of pain and swelling for 1 year, which was unresponsive to antibiotics. The diagnosis of XO was confirmed histopathologically, when the culture of the same tissue grew Mycobacterium marinum. Radical synovectomy of the lesion was performed, after which minocycline, clarithromycin, and ethambutol were administered. In 2012, Borjian and colleagues⁸ reported a case of XO of the proximal humerus and proximal fibula in a 14-year-old child. The child, who presented with fever, pain, and restriction of shoulder movements, was started on oral antibiotics as the tissue culture grew Staphylococcus aureus; the patient did not complete the course of treatment in the hospital. No surgical intervention was done in this case. The diagnosis of XO was confirmed by microscopic examination of the tissue.

An association between bacterial infection and xanthogranulomatous inflammation has existed in several organs, such as the kidneys, and in the gastrointestinal system, but such an association of the 2 is yet to be determined for bone.^5,10,16-19 Because of the paucity of literature on the disease, a management protocol for XO of bone has not been defined, and decisions have to be made considering the natural history of the disease in other organs. We present this case primarily because of its rarity, curability, and its close resemblance to bone tumors. While XO is benign, it can mimic a neoplastic bone lesion in its imaging and clinical manifestations, and appropriate differentiation is crucial. Currently, histopathologic examination of lesions is the most specific and is the gold standard for diagnosis.

Conclusion

Xanthogranulomatous osteomyelitis is a very rare entity, and only a few cases have been reported in the English-language literature. Though rare, XO warrants greater emphasis than it receives in the literature. It is a chronic inflammatory disease having a close resemblance to bone tumors. A high index of suspicion must be practiced to differentiate XO from tumors. Histopathologic examination is mandatory to establish definitive diagnosis and correct treatment.

Xanthogranulomatous osteomyelitis (XO) is a type of chronic inflammatory process that is characterized by the collection of foamy macrophages along with mononuclear cells in the tissue.¹ Xanthogranulomatous osteomyelitis is characterized by the presence of granular, eosinophilic, periodic acid–Schiff–positive histiocytes in the initial stages, followed by the mixture of foamy macrophages and activated plasma cells and, last, by the presence of suppurative foci and hemorrhage. This is an uncommon process best known to occur in the gallbladder, kidney, urinary bladder, fallopian tube, ovary, vagina, prostate, testis, epididymis, colon, and appendix.^2-4 Very rarely, it can affect lungs, brain, or bone. Only 5 cases of XO have been reported in the literature.^5-8

We report XO of the proximal femur in a 65-year-old woman who initially had a clinical and radiologic diagnosis of aneurysmal bone cyst; however, histopathologic examination confirmed the diagnosis of XO. Xanthogranulomatous osteomyelitis mimics a neoplastic pathology in gallbladder, kidney, and prostrate on gross clinical and radiologic examination.⁹ The pathogenesis of XO is best characterized by a delayed type of hypersensitivity reaction.¹⁰ The differential diagnosis includes chronic recurrent multifocal osteomyelitis, xanthoma, infiltrative storage disorder, malakoplakia, Langerhans cell histiocytosis, fibrohistiocytic tumor, Erdheim-Chester disease, and metastatic renal cell carcinoma.^11-14 The patient provided written informed consent for print and electronic publication of this case report.

Case Report

A 65-year-old hypertensive woman presented with complaints of pain in the right hip for a duration of 6 months. Pain was radiating from the right hip region to the anteromedial aspect of the knee and progressively increasing, with a history of pain at rest suggestive of a nonmechanical pathology in the hip. There was no history of fever, weight loss, loss of appetite, pain in any other joint, or morning stiffness. The patient was mobile without support and was able to squat and sit cross-legged; however, the stance phase on the right side was less than on the left side, suggestive of an antalgic component in the gait.

On examining the patient, there was anterior hip joint tenderness with no local sign of any infective or inflammatory pathology. Trochanteric tenderness was present, but there was no irregularity, broadening, or thickening of the trochanter. There was no restriction in the range of motion, and no coronal or sagittal plane deformity in the right hip. There was no limb-length discrepancy. However, the patient was not able to raise her leg actively, probably because of pain in the right hip.

On plain radiographs of the pelvis with bilateral hips, a well-defined nonexpansile uniloculated lytic lesion with sclerotic margins was present in the neck of the right femur, extending to the intertrochanteric area (Figure 1). Ground-glass appearance was also noted. Considering the benign nature of the lesion radiologically and clinically, a differential diagnosis of hyperparathyroidism, renal osteodystrophy, multiple myeloma, and fibrous dysplasia was considered. Hematologic investigations, skeletal survey, and magnetic resonance imaging (MRI) of the bilateral hips were performed to rule out the differential diagnosis.

The patient’s hemoglobin level was 11.8 g/dL with total white blood cell count of 10,300/µL. Renal and hepatic functions were within normal limit. Serum erythrocyte sedimentation rate (ESR) was 12 mm/h and C-reactive protein level was normal. Serum parathyroid level was 32 pg/mL, which was within normal limits, with an alkaline phosphatase level of 101 U/L. The skeletal survey showed no other bony lesion in the body. T1-weighted MRI of both hips showed a well-defined hypointense lesion in the neck and intertrochanteric area of the right hip, which was hyperintense on T2-weighted MRI, suggestive of aneurysmal bone cyst (Figure 2).

Normal ESR, hemoglobin, alkaline phosphatase, and serum parathyroid levels and normal skeletal survey almost ruled out multiple myeloma and hyperparathyroidism. Normal renal profile ruled out renal osteodystrophy and the osteitis fibrosa cystica lesion associated with it. We planned for prophylactic internal fixation of the lesion to prevent a pathologic fracture. According to Mirels,¹⁵ if there is a lytic lesion covering more than two-thirds of the circumference of the bone in the peritrochanteric area, the chances of a pathologic fracture are high and such fractures should be fixed.

We planned for curettage of the lesion with bone grafting and in situ intramedullary fixation of the lesion. Curettage was done according to the plan and the sample was sent for histopathologic examination. In situ internal fixation and bone grafting were performed by using a proximal femoral intramedullary nail. To our surprise, the biopsy sample was reported as xanthogranuloma, with multiple foamy macrophages mixed with inflammatory cells and aggregates of lymphocytes (Figure 3). Mycobacterial and routine bacterial cultures were reported as negative. The patient was kept on oral antibiotics (cefixime and moxifloxacin) for 6 weeks, and she made an uneventful recovery. At 6-month follow-up, a radiograph of the right hip showed a healed lesion with proximal femoral nail in situ (Figure 4).

Discussion

To the best of our knowledge, a total of 5 cases of XO have been reported in the literature. The earliest of these reports were by Cozzutto and Carbone,¹ who reported 2 cases of XO of the first rib and of the epiphysis of the tibia, respectively. The importance of these lesions to diagnosis is their confusion with a neoplastic disease, as XO is itself a benign disorder. These lesions can mimic a neoplastic lesion in clinical and radiologic presentation and the only way to differentiate the lesion from a neoplastic disease is by histopathologic examination of the tissue. Hypothetically, xanthogranulomatous disorders can be related to trauma or infection.

In 2007, Vankalakunti and colleagues⁶ reported XO of the ulna in a 50-year-old postmenopausal woman. In that case, progressive swelling was present on the extensor aspect of her right forearm for a period of 2 years, for which curettage and bone grafting were performed, using autograft from the ipsilateral iliac crest. The tissue culture was sterile, and XO was diagnosed as a result of the histopathologic examination. In 2009, Cennimo and colleagues⁷ reported XO of the index finger and wrist of a man complaining of pain and swelling for 1 year, which was unresponsive to antibiotics. The diagnosis of XO was confirmed histopathologically, when the culture of the same tissue grew Mycobacterium marinum. Radical synovectomy of the lesion was performed, after which minocycline, clarithromycin, and ethambutol were administered. In 2012, Borjian and colleagues⁸ reported a case of XO of the proximal humerus and proximal fibula in a 14-year-old child. The child, who presented with fever, pain, and restriction of shoulder movements, was started on oral antibiotics as the tissue culture grew Staphylococcus aureus; the patient did not complete the course of treatment in the hospital. No surgical intervention was done in this case. The diagnosis of XO was confirmed by microscopic examination of the tissue.

An association between bacterial infection and xanthogranulomatous inflammation has existed in several organs, such as the kidneys, and in the gastrointestinal system, but such an association of the 2 is yet to be determined for bone.^5,10,16-19 Because of the paucity of literature on the disease, a management protocol for XO of bone has not been defined, and decisions have to be made considering the natural history of the disease in other organs. We present this case primarily because of its rarity, curability, and its close resemblance to bone tumors. While XO is benign, it can mimic a neoplastic bone lesion in its imaging and clinical manifestations, and appropriate differentiation is crucial. Currently, histopathologic examination of lesions is the most specific and is the gold standard for diagnosis.

Conclusion

Xanthogranulomatous osteomyelitis is a very rare entity, and only a few cases have been reported in the English-language literature. Though rare, XO warrants greater emphasis than it receives in the literature. It is a chronic inflammatory disease having a close resemblance to bone tumors. A high index of suspicion must be practiced to differentiate XO from tumors. Histopathologic examination is mandatory to establish definitive diagnosis and correct treatment.

Gamekeeper’s or skier’s thumb is caused by an injury to the ulnar collateral ligament (UCL) of the metacarpophalangeal (MCP) joint of the thumb. The mechanism of injury is forced radial and palmar abduction and hyperextension.

This lesion was initially described in 1955 by Campbell.¹ It occurred in gamekeepers who worked in preserves in Scotland. The UCL was injured because of the way they killed rabbits—hence, gamekeeper’s thumb. Now these injuries are more common in skiers—skier’s thumb. In skiers, the mechanism of injury is the force exerted by the ski pole strap on the thumb during a fall. This injury is also seen in breakdancers.^1,2

Different lesions can result, the most common being that of the UCL. The UCL lesion may be partial, with no joint instability,^3,4 or total, with instability and subdislocation of the proximal phalanx.^5-9 Rupture of the thumb adductor aponeurosis and displacement of the long extensor have been described as the cause of thumb instability.^6-8

UCL rupture can occur in its extension or can cause a fracture-tearing in the proximal phalanx.^9-12 Intra-articular fractures are sometimes found. The essential problem in UCL injuries is the impossibility of spontaneous healing once the rupture is complete, because of the Stener effect. (When the UCL ruptures, its proximal part retracts and runs above the fibrous expansion of the adductor muscle, which is interposed between the 2 parts of the ruptured UCL and prevents healing, even if the thumb is immobilized.) In these cases, only surgery can repair the lesion.²

In any thumb injury, particularly one caused by hyperabduction, a UCL lesion should be considered. The main problem is diagnosing sprain severity, which is evidenced by the degree of joint hypermobility. Radiologic examination should be performed in all cases to rule out fracture with tear, posterior capsular tear, palmar plate tear, and palmar subdislocation of the proximal phalanx, all of which are associated with UCL tearing.^7-9

If the diagnosis is suspected, and radiographs show no fracture, comparative radiographs should be obtained in forced valgus.

Technique

We report on a simple, reliable, reproducible method that allows the patient’s thumbs to be compared, under the same force application conditions, on a single radiograph. This technique reduces the patient’s and examiner’s exposure to x-rays and is well tolerated by the patient. Anesthesia for the thumb is usually not necessary.

In each hand, the patient holds a cylindrical object, such as a drinking glass (standard diameter, 7.5-8.5 cm). We use an elastic crepe bandage roll (diameter, 7.5 cm; width, 10 cm). This roll is common in emergency departments (EDs) and easily accessible. The patient holds the rolls in his or her hands with the thumbs in the posteroanterior position (Figures 1–3) and places himself or herself on a 18×24-cm frame or directly on the radiography table.

Both thumbs are captured on a single functional radiograph for comparison of forced valgus of the MCP joints, as in our example cases. The patients provided written informed consent for print and electronic publication of these case reports.

Case Reports

Control Case

The single functional radiograph of both thumbs showed no evidence of joint laxity on the valgus stress test (Figure 4).

Case 1

A 72-year-old woman landed on her left hand when she fell backward while supporting the hand on a piece of furniture. She presented to the ED with pain in the region of the thumb and thenar eminence. Posteroanterior and lateral radiograph projections showed no significant bone injury (Figure 5). Given the patient’s persistent pain, the traumatologist suspected damage to the thumb UCL, so a simultaneous bilateral functional radiographic projection was obtained. The projection showed joint laxity, implying damage to the thumb UCL. Repair and reinsertion of the UCL were performed using a bone harpoon suture.

Case 2

A 58-year-old man sustained a left hand injury when, using both hands, he tried to catch hold of a falling wooden plank. When he presented to the ED the following week, he was given a diagnosis of thumb contusion and forced hyperabduction and was wearing a metal strap for immobilization. Radiographs showed no bone damage (Figure 6). Thumb UCL injury was suspected on the basis of the physical examination findings and the mechanism of injury. A bilateral simultaneous functional radiographic projection showed significant joint laxity. Surgical treatment with the pull-out technique was performed.

Case 3

A 44-year-old woman experienced forced traction from a dog leash and presented to the ED with pain in the right thumb region. Radiographs showed no bone damage (Figure 7). Thumb UCL injury was suspected. A bilateral simultaneous functional radiographic projection showed slight joint laxity, a sprain was diagnosed, and plaster bandaging was applied. Figures 8A–8D show the accurate thumb positions for performing the functional radiograph in forced valgus. We call the technique J.J.’s thumb radiographic projection.

Discussion

Examination using the stress test to cause joint tilt is crucial in making an accurate diagnosis and deciding on the most appropriate therapeutic approach.¹⁰ Most authors accept that surgical management is required in joint tilts over 30º, as these involve complete UCL rupture.^10-12

The MCP joint must be examined in flexion, when the main fascicle of the UCL is tight, and not in extension, when the main fascicle of the UCL is relaxed. If we examine the thumb in extension, radial deviations may occur that are not caused by joint instability. Tilt here must be compared with that of the healthy side.¹¹

Early diagnosis and adequate management are essential, as unnoticed or undervalued injuries can progress to painful sequelae, associated with stiffness, instability, and osteoarthritis, with evident harm to the grip and pinch functions of the hand. In many cases, clinical evidence of MCP joint instability is difficult. The radiologic diagnosis is usually obtained with comparative radiographs in forced valgus of both thumbs.

The forced valgus maneuver typically is performed by the examiner, who must stay with the patient in the radiography room and wear radiologic protection. Incredibly, some patients must force the valgus themselves.

The maneuver we have described clearly has complications, as it is painful, and some patients are uncooperative. Usually the thumb is anesthetized, and the examiner assumes the exposure to x-rays. The valgus deviation force that can be applied during stability testing may lead to further disruption of a partially torn ligament or displacement of a ruptured ligament if the overforced maneuver is performed.^13,14 That does not occur with our technique. On the other hand, the forces applied to the thumbs must be symmetrical for comparison purposes. The way to prevent these inconveniences is to perform the forced valgus maneuver over both thumbs simultaneously, under the same force application conditions and on a single radiograph, without requiring the examiner to remain with the patient in the radiography room.

Heim¹⁵ designed a system for simultaneous functional radiographs, but an apparatus must be built to adapt it to the frame of the radiography table, and the technique involves hyperpronating both hands and bandaging them to the forearm—which is uncomfortable and bothersome for patients and, in our opinion, has a poor application in high-volume EDs.

The technique of having the patient hold a bandage roll (J.J.’s thumb radiographic projection) offers several advantages:

1.  The thumb can be placed in flexion, tightening the main fascicle of the UCL, which is how the UCL must be examined.

2. Forced valgus is allowed. Holding a water glass involves opening the thumb and the necessary stability of the MCP joint of the thumb (grip function of thumb); this radiographic technique is functional.

3. The examiner need not stay with the patient in the radiography room or be exposed to x-rays.

4. The bandage roll is thick enough to generate forced valgus in a patient with large hands. The nonrigid roll makes the examination more tolerable and avoids overforced valgus, eliminating the need for anesthetic blockade.

5. The technique is accessible and simple. In fact, there is no need to remove the roll from its wrapping.

Gamekeeper’s or skier’s thumb is caused by an injury to the ulnar collateral ligament (UCL) of the metacarpophalangeal (MCP) joint of the thumb. The mechanism of injury is forced radial and palmar abduction and hyperextension.

This lesion was initially described in 1955 by Campbell.¹ It occurred in gamekeepers who worked in preserves in Scotland. The UCL was injured because of the way they killed rabbits—hence, gamekeeper’s thumb. Now these injuries are more common in skiers—skier’s thumb. In skiers, the mechanism of injury is the force exerted by the ski pole strap on the thumb during a fall. This injury is also seen in breakdancers.^1,2

Different lesions can result, the most common being that of the UCL. The UCL lesion may be partial, with no joint instability,^3,4 or total, with instability and subdislocation of the proximal phalanx.^5-9 Rupture of the thumb adductor aponeurosis and displacement of the long extensor have been described as the cause of thumb instability.^6-8

UCL rupture can occur in its extension or can cause a fracture-tearing in the proximal phalanx.^9-12 Intra-articular fractures are sometimes found. The essential problem in UCL injuries is the impossibility of spontaneous healing once the rupture is complete, because of the Stener effect. (When the UCL ruptures, its proximal part retracts and runs above the fibrous expansion of the adductor muscle, which is interposed between the 2 parts of the ruptured UCL and prevents healing, even if the thumb is immobilized.) In these cases, only surgery can repair the lesion.²

In any thumb injury, particularly one caused by hyperabduction, a UCL lesion should be considered. The main problem is diagnosing sprain severity, which is evidenced by the degree of joint hypermobility. Radiologic examination should be performed in all cases to rule out fracture with tear, posterior capsular tear, palmar plate tear, and palmar subdislocation of the proximal phalanx, all of which are associated with UCL tearing.^7-9

If the diagnosis is suspected, and radiographs show no fracture, comparative radiographs should be obtained in forced valgus.

Technique

We report on a simple, reliable, reproducible method that allows the patient’s thumbs to be compared, under the same force application conditions, on a single radiograph. This technique reduces the patient’s and examiner’s exposure to x-rays and is well tolerated by the patient. Anesthesia for the thumb is usually not necessary.

In each hand, the patient holds a cylindrical object, such as a drinking glass (standard diameter, 7.5-8.5 cm). We use an elastic crepe bandage roll (diameter, 7.5 cm; width, 10 cm). This roll is common in emergency departments (EDs) and easily accessible. The patient holds the rolls in his or her hands with the thumbs in the posteroanterior position (Figures 1–3) and places himself or herself on a 18×24-cm frame or directly on the radiography table.

Both thumbs are captured on a single functional radiograph for comparison of forced valgus of the MCP joints, as in our example cases. The patients provided written informed consent for print and electronic publication of these case reports.

Case Reports

Control Case

The single functional radiograph of both thumbs showed no evidence of joint laxity on the valgus stress test (Figure 4).

Case 1

A 72-year-old woman landed on her left hand when she fell backward while supporting the hand on a piece of furniture. She presented to the ED with pain in the region of the thumb and thenar eminence. Posteroanterior and lateral radiograph projections showed no significant bone injury (Figure 5). Given the patient’s persistent pain, the traumatologist suspected damage to the thumb UCL, so a simultaneous bilateral functional radiographic projection was obtained. The projection showed joint laxity, implying damage to the thumb UCL. Repair and reinsertion of the UCL were performed using a bone harpoon suture.

Case 2

A 58-year-old man sustained a left hand injury when, using both hands, he tried to catch hold of a falling wooden plank. When he presented to the ED the following week, he was given a diagnosis of thumb contusion and forced hyperabduction and was wearing a metal strap for immobilization. Radiographs showed no bone damage (Figure 6). Thumb UCL injury was suspected on the basis of the physical examination findings and the mechanism of injury. A bilateral simultaneous functional radiographic projection showed significant joint laxity. Surgical treatment with the pull-out technique was performed.

Case 3

A 44-year-old woman experienced forced traction from a dog leash and presented to the ED with pain in the right thumb region. Radiographs showed no bone damage (Figure 7). Thumb UCL injury was suspected. A bilateral simultaneous functional radiographic projection showed slight joint laxity, a sprain was diagnosed, and plaster bandaging was applied. Figures 8A–8D show the accurate thumb positions for performing the functional radiograph in forced valgus. We call the technique J.J.’s thumb radiographic projection.

Discussion

Examination using the stress test to cause joint tilt is crucial in making an accurate diagnosis and deciding on the most appropriate therapeutic approach.¹⁰ Most authors accept that surgical management is required in joint tilts over 30º, as these involve complete UCL rupture.^10-12

The MCP joint must be examined in flexion, when the main fascicle of the UCL is tight, and not in extension, when the main fascicle of the UCL is relaxed. If we examine the thumb in extension, radial deviations may occur that are not caused by joint instability. Tilt here must be compared with that of the healthy side.¹¹

Early diagnosis and adequate management are essential, as unnoticed or undervalued injuries can progress to painful sequelae, associated with stiffness, instability, and osteoarthritis, with evident harm to the grip and pinch functions of the hand. In many cases, clinical evidence of MCP joint instability is difficult. The radiologic diagnosis is usually obtained with comparative radiographs in forced valgus of both thumbs.

The forced valgus maneuver typically is performed by the examiner, who must stay with the patient in the radiography room and wear radiologic protection. Incredibly, some patients must force the valgus themselves.

The maneuver we have described clearly has complications, as it is painful, and some patients are uncooperative. Usually the thumb is anesthetized, and the examiner assumes the exposure to x-rays. The valgus deviation force that can be applied during stability testing may lead to further disruption of a partially torn ligament or displacement of a ruptured ligament if the overforced maneuver is performed.^13,14 That does not occur with our technique. On the other hand, the forces applied to the thumbs must be symmetrical for comparison purposes. The way to prevent these inconveniences is to perform the forced valgus maneuver over both thumbs simultaneously, under the same force application conditions and on a single radiograph, without requiring the examiner to remain with the patient in the radiography room.

Heim¹⁵ designed a system for simultaneous functional radiographs, but an apparatus must be built to adapt it to the frame of the radiography table, and the technique involves hyperpronating both hands and bandaging them to the forearm—which is uncomfortable and bothersome for patients and, in our opinion, has a poor application in high-volume EDs.

The technique of having the patient hold a bandage roll (J.J.’s thumb radiographic projection) offers several advantages:

1.  The thumb can be placed in flexion, tightening the main fascicle of the UCL, which is how the UCL must be examined.

2. Forced valgus is allowed. Holding a water glass involves opening the thumb and the necessary stability of the MCP joint of the thumb (grip function of thumb); this radiographic technique is functional.

3. The examiner need not stay with the patient in the radiography room or be exposed to x-rays.

4. The bandage roll is thick enough to generate forced valgus in a patient with large hands. The nonrigid roll makes the examination more tolerable and avoids overforced valgus, eliminating the need for anesthetic blockade.

5. The technique is accessible and simple. In fact, there is no need to remove the roll from its wrapping.

Gamekeeper’s or skier’s thumb is caused by an injury to the ulnar collateral ligament (UCL) of the metacarpophalangeal (MCP) joint of the thumb. The mechanism of injury is forced radial and palmar abduction and hyperextension.

This lesion was initially described in 1955 by Campbell.¹ It occurred in gamekeepers who worked in preserves in Scotland. The UCL was injured because of the way they killed rabbits—hence, gamekeeper’s thumb. Now these injuries are more common in skiers—skier’s thumb. In skiers, the mechanism of injury is the force exerted by the ski pole strap on the thumb during a fall. This injury is also seen in breakdancers.^1,2

Different lesions can result, the most common being that of the UCL. The UCL lesion may be partial, with no joint instability,^3,4 or total, with instability and subdislocation of the proximal phalanx.^5-9 Rupture of the thumb adductor aponeurosis and displacement of the long extensor have been described as the cause of thumb instability.^6-8

UCL rupture can occur in its extension or can cause a fracture-tearing in the proximal phalanx.^9-12 Intra-articular fractures are sometimes found. The essential problem in UCL injuries is the impossibility of spontaneous healing once the rupture is complete, because of the Stener effect. (When the UCL ruptures, its proximal part retracts and runs above the fibrous expansion of the adductor muscle, which is interposed between the 2 parts of the ruptured UCL and prevents healing, even if the thumb is immobilized.) In these cases, only surgery can repair the lesion.²

In any thumb injury, particularly one caused by hyperabduction, a UCL lesion should be considered. The main problem is diagnosing sprain severity, which is evidenced by the degree of joint hypermobility. Radiologic examination should be performed in all cases to rule out fracture with tear, posterior capsular tear, palmar plate tear, and palmar subdislocation of the proximal phalanx, all of which are associated with UCL tearing.^7-9

If the diagnosis is suspected, and radiographs show no fracture, comparative radiographs should be obtained in forced valgus.

Technique

We report on a simple, reliable, reproducible method that allows the patient’s thumbs to be compared, under the same force application conditions, on a single radiograph. This technique reduces the patient’s and examiner’s exposure to x-rays and is well tolerated by the patient. Anesthesia for the thumb is usually not necessary.

In each hand, the patient holds a cylindrical object, such as a drinking glass (standard diameter, 7.5-8.5 cm). We use an elastic crepe bandage roll (diameter, 7.5 cm; width, 10 cm). This roll is common in emergency departments (EDs) and easily accessible. The patient holds the rolls in his or her hands with the thumbs in the posteroanterior position (Figures 1–3) and places himself or herself on a 18×24-cm frame or directly on the radiography table.

Both thumbs are captured on a single functional radiograph for comparison of forced valgus of the MCP joints, as in our example cases. The patients provided written informed consent for print and electronic publication of these case reports.

Case Reports

Control Case

The single functional radiograph of both thumbs showed no evidence of joint laxity on the valgus stress test (Figure 4).

Case 1

A 72-year-old woman landed on her left hand when she fell backward while supporting the hand on a piece of furniture. She presented to the ED with pain in the region of the thumb and thenar eminence. Posteroanterior and lateral radiograph projections showed no significant bone injury (Figure 5). Given the patient’s persistent pain, the traumatologist suspected damage to the thumb UCL, so a simultaneous bilateral functional radiographic projection was obtained. The projection showed joint laxity, implying damage to the thumb UCL. Repair and reinsertion of the UCL were performed using a bone harpoon suture.

Case 2

A 58-year-old man sustained a left hand injury when, using both hands, he tried to catch hold of a falling wooden plank. When he presented to the ED the following week, he was given a diagnosis of thumb contusion and forced hyperabduction and was wearing a metal strap for immobilization. Radiographs showed no bone damage (Figure 6). Thumb UCL injury was suspected on the basis of the physical examination findings and the mechanism of injury. A bilateral simultaneous functional radiographic projection showed significant joint laxity. Surgical treatment with the pull-out technique was performed.

Case 3

A 44-year-old woman experienced forced traction from a dog leash and presented to the ED with pain in the right thumb region. Radiographs showed no bone damage (Figure 7). Thumb UCL injury was suspected. A bilateral simultaneous functional radiographic projection showed slight joint laxity, a sprain was diagnosed, and plaster bandaging was applied. Figures 8A–8D show the accurate thumb positions for performing the functional radiograph in forced valgus. We call the technique J.J.’s thumb radiographic projection.

Discussion

Examination using the stress test to cause joint tilt is crucial in making an accurate diagnosis and deciding on the most appropriate therapeutic approach.¹⁰ Most authors accept that surgical management is required in joint tilts over 30º, as these involve complete UCL rupture.^10-12

The MCP joint must be examined in flexion, when the main fascicle of the UCL is tight, and not in extension, when the main fascicle of the UCL is relaxed. If we examine the thumb in extension, radial deviations may occur that are not caused by joint instability. Tilt here must be compared with that of the healthy side.¹¹

Early diagnosis and adequate management are essential, as unnoticed or undervalued injuries can progress to painful sequelae, associated with stiffness, instability, and osteoarthritis, with evident harm to the grip and pinch functions of the hand. In many cases, clinical evidence of MCP joint instability is difficult. The radiologic diagnosis is usually obtained with comparative radiographs in forced valgus of both thumbs.

The forced valgus maneuver typically is performed by the examiner, who must stay with the patient in the radiography room and wear radiologic protection. Incredibly, some patients must force the valgus themselves.

The maneuver we have described clearly has complications, as it is painful, and some patients are uncooperative. Usually the thumb is anesthetized, and the examiner assumes the exposure to x-rays. The valgus deviation force that can be applied during stability testing may lead to further disruption of a partially torn ligament or displacement of a ruptured ligament if the overforced maneuver is performed.^13,14 That does not occur with our technique. On the other hand, the forces applied to the thumbs must be symmetrical for comparison purposes. The way to prevent these inconveniences is to perform the forced valgus maneuver over both thumbs simultaneously, under the same force application conditions and on a single radiograph, without requiring the examiner to remain with the patient in the radiography room.

Heim¹⁵ designed a system for simultaneous functional radiographs, but an apparatus must be built to adapt it to the frame of the radiography table, and the technique involves hyperpronating both hands and bandaging them to the forearm—which is uncomfortable and bothersome for patients and, in our opinion, has a poor application in high-volume EDs.

The technique of having the patient hold a bandage roll (J.J.’s thumb radiographic projection) offers several advantages:

1.  The thumb can be placed in flexion, tightening the main fascicle of the UCL, which is how the UCL must be examined.

2. Forced valgus is allowed. Holding a water glass involves opening the thumb and the necessary stability of the MCP joint of the thumb (grip function of thumb); this radiographic technique is functional.

3. The examiner need not stay with the patient in the radiography room or be exposed to x-rays.

4. The bandage roll is thick enough to generate forced valgus in a patient with large hands. The nonrigid roll makes the examination more tolerable and avoids overforced valgus, eliminating the need for anesthetic blockade.

5. The technique is accessible and simple. In fact, there is no need to remove the roll from its wrapping.

Extensor tendon rupture in chronic Madelung deformity, as a result of tendon attrition on the dislocated distal ulna, occurs infrequently. However, it is often seen in patients with rheumatoid arthritis. This issue has been reported in only a few English-language case reports. Here we report a case of multiple tendon ruptures in a previously undiagnosed Madelung deformity. The patient provided written informed consent for print and electronic publication of this case report.

Case Report

A 56-year-old active woman presented with 50 days’ inability to extend the fourth and fifth fingers of her dominant right hand. The loss of finger extension progressed, over several weeks, to involve the third finger as well. The first 2 tendon ruptures had been triggered by lifting a light grocery bag, when she noticed a sharp sudden pain and “pop.” The third rupture occurred spontaneously with a snapping sound the night before surgery.

 The patient had observed some prominence on the ulnar side of her right wrist since childhood but had never experienced any pain or functional disability. There was neither history of trauma, inflammatory disease, diabetes mellitus, or infection, nor positive family history of similar wrist deformity.

The physical examination showed a dorsally subluxated distal radioulnar joint, prominent ulnar styloid, and mild ulnar and volar deviation of the wrist along with limitation of wrist dorsiflexion. Complete loss of active extension of the 3 ulnar fingers was demonstrated, while neurovascular status and all other hand evaluations were normal. The wrist radiographs confirmed the typical findings of Madelung deformity (Figure 1).

Repair of the ruptured tendons and resection of the prominent distal ulna (Darrach procedure) was planned. (Given the patient’s age and evidence of degenerative changes in the radiocarpal joint, correction of the Madelung deformity did not seem necessary). At time of surgery, the recently ruptured third finger extensor tendon was easily found and approximated, and end-to-end repair was performed. The fourth and fifth fingers, however, had to be fished out more proximally from dense granulation tissue. After the distal ulna was resected for a distance of 1.5 cm, meticulous repair of the ulnar collateral ligament and the capsule and periosteum over the end of the ulna was performed. Then, for grafting of the ruptured tendons, the extensor indicis proprius tendon was isolated and transected at the second metacarpophalangeal joint level. A piece of this tendon was used as interpositional graft for the fourth extensor tendon, and the main tendon unit was transferred to the fifth finger extensor. The extensor digiti quinti tendon, which was about to rupture, was further reinforced by suturing it side to side to the muscle and tendon of the extensor indicis proprius (Figure 2).

Postoperatively, the wrist was kept in extension in a cast for 3 weeks while the fingers were free for active movement. A removable wrist splint was used for an additional month. At 3-month follow-up, the patient had regained full and strong finger extension and wrist motion.

At 3-year follow-up, the patient was pain-free, and had full extension of all fingers, full forearm rotation, and near-normal motion (better than her preoperative motion). The grip power on the operated right hand was 215 N, and pinch power was 93 N. (The values for the left side were 254 N and 83 N, respectively, using the Jamar hydraulic hand dynamometer [Patterson Medical].) The patient has had no additional tendon rupture (Figure 3).

Discussion

Madelung deformity was first described by Madelung in 1878 and several cases have reported this deformity. However, extensor tendon rupture caused by Madelung deformity is very rare, reported in few cases.¹

Extensor tendon rupture caused by chronic Madelung deformity has been reported few times in the English literature. Goodwin¹ apparently published the first report of such an occurrence in 1979. Ducloyer and colleagues² from France reported 6 cases of extensor tendon rupture as a result of inferior distal radioulnar joint deformity of Madelung. Jebson and colleagues³ reported bilateral spontaneous extensor tendon ruptures in Madelung deformity in 1992.

The mechanism of tendon rupture seems to be mechanical, resulting from continuous rubbing and erosion of tendons over the deformed ulnar head, which has a rough irregular surface⁴ and leads to fraying of the tendons and eventual rupture and retraction of the severed tendon ends. This rupture usually progresses stepwise from more medial to the lateral tendons.² Older patients are, therefore, subject to chronic repetitive attritional trauma leading to tendon rupture.

Tendons may rupture as a result of a variety of conditions, such as chronic synovitis in rheumatoid arthritis, systemic lupus erythematosus, mixed connective tissue disease, or crystal deposition in gout.^5-8 Some other metabolic or endocrine conditions that involve tendon ruptures include diabetes mellitus, chronic renal failure, and hyperparathyroidism. Steroid injection into the tendons also has a detrimental effect on tendon integrity and may cause tendon tear.⁹ Mechanical factors, such as erosion on bony prominences, are well-known etiologies for tendon rupture, as commonly seen in rheumatoid arthritis, and have been reported in Kienböck disease,¹⁰ thumb carpometacarpal arthritis,¹¹ Colles fracture, scaphoid fracture nonunion,¹² and Madelung deformity.

Conclusion

Our case reflects the usual middle-aged female presentation of such a tendon rupture. The tendon ruptures were spontaneous in the reported order of ulnar to radial, beginning with the little and ring fingers, and progressed radially. The patient had isolated Madelung deformity with no other sign of dyschondrosteosis¹³ or dwarfism, conditions commonly mentioned in association with Madelung deformity. This case report should raise awareness about possible tendon rupture in any chronic case of Madelung deformity.

Extensor tendon rupture in chronic Madelung deformity, as a result of tendon attrition on the dislocated distal ulna, occurs infrequently. However, it is often seen in patients with rheumatoid arthritis. This issue has been reported in only a few English-language case reports. Here we report a case of multiple tendon ruptures in a previously undiagnosed Madelung deformity. The patient provided written informed consent for print and electronic publication of this case report.

Case Report

A 56-year-old active woman presented with 50 days’ inability to extend the fourth and fifth fingers of her dominant right hand. The loss of finger extension progressed, over several weeks, to involve the third finger as well. The first 2 tendon ruptures had been triggered by lifting a light grocery bag, when she noticed a sharp sudden pain and “pop.” The third rupture occurred spontaneously with a snapping sound the night before surgery.

 The patient had observed some prominence on the ulnar side of her right wrist since childhood but had never experienced any pain or functional disability. There was neither history of trauma, inflammatory disease, diabetes mellitus, or infection, nor positive family history of similar wrist deformity.

The physical examination showed a dorsally subluxated distal radioulnar joint, prominent ulnar styloid, and mild ulnar and volar deviation of the wrist along with limitation of wrist dorsiflexion. Complete loss of active extension of the 3 ulnar fingers was demonstrated, while neurovascular status and all other hand evaluations were normal. The wrist radiographs confirmed the typical findings of Madelung deformity (Figure 1).

Repair of the ruptured tendons and resection of the prominent distal ulna (Darrach procedure) was planned. (Given the patient’s age and evidence of degenerative changes in the radiocarpal joint, correction of the Madelung deformity did not seem necessary). At time of surgery, the recently ruptured third finger extensor tendon was easily found and approximated, and end-to-end repair was performed. The fourth and fifth fingers, however, had to be fished out more proximally from dense granulation tissue. After the distal ulna was resected for a distance of 1.5 cm, meticulous repair of the ulnar collateral ligament and the capsule and periosteum over the end of the ulna was performed. Then, for grafting of the ruptured tendons, the extensor indicis proprius tendon was isolated and transected at the second metacarpophalangeal joint level. A piece of this tendon was used as interpositional graft for the fourth extensor tendon, and the main tendon unit was transferred to the fifth finger extensor. The extensor digiti quinti tendon, which was about to rupture, was further reinforced by suturing it side to side to the muscle and tendon of the extensor indicis proprius (Figure 2).

Postoperatively, the wrist was kept in extension in a cast for 3 weeks while the fingers were free for active movement. A removable wrist splint was used for an additional month. At 3-month follow-up, the patient had regained full and strong finger extension and wrist motion.

At 3-year follow-up, the patient was pain-free, and had full extension of all fingers, full forearm rotation, and near-normal motion (better than her preoperative motion). The grip power on the operated right hand was 215 N, and pinch power was 93 N. (The values for the left side were 254 N and 83 N, respectively, using the Jamar hydraulic hand dynamometer [Patterson Medical].) The patient has had no additional tendon rupture (Figure 3).

Discussion

Madelung deformity was first described by Madelung in 1878 and several cases have reported this deformity. However, extensor tendon rupture caused by Madelung deformity is very rare, reported in few cases.¹

Extensor tendon rupture caused by chronic Madelung deformity has been reported few times in the English literature. Goodwin¹ apparently published the first report of such an occurrence in 1979. Ducloyer and colleagues² from France reported 6 cases of extensor tendon rupture as a result of inferior distal radioulnar joint deformity of Madelung. Jebson and colleagues³ reported bilateral spontaneous extensor tendon ruptures in Madelung deformity in 1992.

The mechanism of tendon rupture seems to be mechanical, resulting from continuous rubbing and erosion of tendons over the deformed ulnar head, which has a rough irregular surface⁴ and leads to fraying of the tendons and eventual rupture and retraction of the severed tendon ends. This rupture usually progresses stepwise from more medial to the lateral tendons.² Older patients are, therefore, subject to chronic repetitive attritional trauma leading to tendon rupture.

Tendons may rupture as a result of a variety of conditions, such as chronic synovitis in rheumatoid arthritis, systemic lupus erythematosus, mixed connective tissue disease, or crystal deposition in gout.^5-8 Some other metabolic or endocrine conditions that involve tendon ruptures include diabetes mellitus, chronic renal failure, and hyperparathyroidism. Steroid injection into the tendons also has a detrimental effect on tendon integrity and may cause tendon tear.⁹ Mechanical factors, such as erosion on bony prominences, are well-known etiologies for tendon rupture, as commonly seen in rheumatoid arthritis, and have been reported in Kienböck disease,¹⁰ thumb carpometacarpal arthritis,¹¹ Colles fracture, scaphoid fracture nonunion,¹² and Madelung deformity.

Conclusion

Our case reflects the usual middle-aged female presentation of such a tendon rupture. The tendon ruptures were spontaneous in the reported order of ulnar to radial, beginning with the little and ring fingers, and progressed radially. The patient had isolated Madelung deformity with no other sign of dyschondrosteosis¹³ or dwarfism, conditions commonly mentioned in association with Madelung deformity. This case report should raise awareness about possible tendon rupture in any chronic case of Madelung deformity.

Extensor tendon rupture in chronic Madelung deformity, as a result of tendon attrition on the dislocated distal ulna, occurs infrequently. However, it is often seen in patients with rheumatoid arthritis. This issue has been reported in only a few English-language case reports. Here we report a case of multiple tendon ruptures in a previously undiagnosed Madelung deformity. The patient provided written informed consent for print and electronic publication of this case report.

Case Report

A 56-year-old active woman presented with 50 days’ inability to extend the fourth and fifth fingers of her dominant right hand. The loss of finger extension progressed, over several weeks, to involve the third finger as well. The first 2 tendon ruptures had been triggered by lifting a light grocery bag, when she noticed a sharp sudden pain and “pop.” The third rupture occurred spontaneously with a snapping sound the night before surgery.

 The patient had observed some prominence on the ulnar side of her right wrist since childhood but had never experienced any pain or functional disability. There was neither history of trauma, inflammatory disease, diabetes mellitus, or infection, nor positive family history of similar wrist deformity.

The physical examination showed a dorsally subluxated distal radioulnar joint, prominent ulnar styloid, and mild ulnar and volar deviation of the wrist along with limitation of wrist dorsiflexion. Complete loss of active extension of the 3 ulnar fingers was demonstrated, while neurovascular status and all other hand evaluations were normal. The wrist radiographs confirmed the typical findings of Madelung deformity (Figure 1).

Repair of the ruptured tendons and resection of the prominent distal ulna (Darrach procedure) was planned. (Given the patient’s age and evidence of degenerative changes in the radiocarpal joint, correction of the Madelung deformity did not seem necessary). At time of surgery, the recently ruptured third finger extensor tendon was easily found and approximated, and end-to-end repair was performed. The fourth and fifth fingers, however, had to be fished out more proximally from dense granulation tissue. After the distal ulna was resected for a distance of 1.5 cm, meticulous repair of the ulnar collateral ligament and the capsule and periosteum over the end of the ulna was performed. Then, for grafting of the ruptured tendons, the extensor indicis proprius tendon was isolated and transected at the second metacarpophalangeal joint level. A piece of this tendon was used as interpositional graft for the fourth extensor tendon, and the main tendon unit was transferred to the fifth finger extensor. The extensor digiti quinti tendon, which was about to rupture, was further reinforced by suturing it side to side to the muscle and tendon of the extensor indicis proprius (Figure 2).

Postoperatively, the wrist was kept in extension in a cast for 3 weeks while the fingers were free for active movement. A removable wrist splint was used for an additional month. At 3-month follow-up, the patient had regained full and strong finger extension and wrist motion.

At 3-year follow-up, the patient was pain-free, and had full extension of all fingers, full forearm rotation, and near-normal motion (better than her preoperative motion). The grip power on the operated right hand was 215 N, and pinch power was 93 N. (The values for the left side were 254 N and 83 N, respectively, using the Jamar hydraulic hand dynamometer [Patterson Medical].) The patient has had no additional tendon rupture (Figure 3).

Discussion

Madelung deformity was first described by Madelung in 1878 and several cases have reported this deformity. However, extensor tendon rupture caused by Madelung deformity is very rare, reported in few cases.¹

Extensor tendon rupture caused by chronic Madelung deformity has been reported few times in the English literature. Goodwin¹ apparently published the first report of such an occurrence in 1979. Ducloyer and colleagues² from France reported 6 cases of extensor tendon rupture as a result of inferior distal radioulnar joint deformity of Madelung. Jebson and colleagues³ reported bilateral spontaneous extensor tendon ruptures in Madelung deformity in 1992.

The mechanism of tendon rupture seems to be mechanical, resulting from continuous rubbing and erosion of tendons over the deformed ulnar head, which has a rough irregular surface⁴ and leads to fraying of the tendons and eventual rupture and retraction of the severed tendon ends. This rupture usually progresses stepwise from more medial to the lateral tendons.² Older patients are, therefore, subject to chronic repetitive attritional trauma leading to tendon rupture.

Tendons may rupture as a result of a variety of conditions, such as chronic synovitis in rheumatoid arthritis, systemic lupus erythematosus, mixed connective tissue disease, or crystal deposition in gout.^5-8 Some other metabolic or endocrine conditions that involve tendon ruptures include diabetes mellitus, chronic renal failure, and hyperparathyroidism. Steroid injection into the tendons also has a detrimental effect on tendon integrity and may cause tendon tear.⁹ Mechanical factors, such as erosion on bony prominences, are well-known etiologies for tendon rupture, as commonly seen in rheumatoid arthritis, and have been reported in Kienböck disease,¹⁰ thumb carpometacarpal arthritis,¹¹ Colles fracture, scaphoid fracture nonunion,¹² and Madelung deformity.

Conclusion

Our case reflects the usual middle-aged female presentation of such a tendon rupture. The tendon ruptures were spontaneous in the reported order of ulnar to radial, beginning with the little and ring fingers, and progressed radially. The patient had isolated Madelung deformity with no other sign of dyschondrosteosis¹³ or dwarfism, conditions commonly mentioned in association with Madelung deformity. This case report should raise awareness about possible tendon rupture in any chronic case of Madelung deformity.