Case Reports

Ewing sarcomas are characterized as primitive malignant round cell tumors.¹ These tumors are diagnosed by neuroectodermal differentiation and by their common histologic and immunohistochemical properties.² Ewing sarcoma is the second most common malignant bone tumor in adolescents and young adults. It is the fourth most common primary malignant tumor, accounting for about 9% of all malignant tumors of bone. The most common primary bone tumors are multiple myeloma, osteosarcoma, and chondrosarcoma.³

The diaphyses of long bones (eg, femur, tibia, humerus) and flat bones (eg, pelvis, scapula) are the most commonly involved sites. Involvement of bones in the hands and feet is uncommon (3%-5% of reported cases).⁴ The foot bones most commonly involved include the calcaneus and the metatarsals, in the series by Casadei and colleagues.⁵

About 90% of Ewing sarcoma cases present before age 20 years (mean age, 13 years).⁶ Typical presentation is that of localized pain at the involved site. Some patients have systemic symptoms, such as fever, malaise, weight loss, leukocytosis, and increased erythrocyte sedimentation rate (ESR) mimicking infection. Radiographically, Ewing sarcoma appears as a permeative destructive bone lesion with a moth-eaten appearance (almost 76% of cases).⁷ This is usually associated with lamellated periosteal new bone formation or an “onion skin” appearance. Less commonly, a sunburst configuration with an associated soft-tissue mass can be seen. Computed tomography (CT) and magnetic resonance imaging (MRI) show the osseous extent of the tumor and the presence or absence of the soft-tissue component of the tumor. Radionuclide bone scans show increased technetium-99m methylene diphosphonate accumulation and are typically hot.⁶

Histopathologically, the tumor is composed of small, uniformly sized cells characterized by an almost clear eosinophilic cytoplasm and very little intercellular matrix. There are lobules and strands divided by prominent septa. Macroscopically, appearance can range from a soft, fleshy solid mass to an almost liquid form, as the lesion does not produce any matrix. At time of surgery, the tumor may have a liquefied component and the appearance of pus.⁶ Prognostic factors are tumor site in foot and treatment according to the series by Casadei and colleagues.⁵ Patients with large central tumors, especially in the pelvis, have worse outcomes than patients with distal tumors.⁸

In this article, we report a case of multifocal Ewing sarcoma involving multiple bones in the foot. Given the multifocal nature of the disease confined to the foot, the initial impression was that of osteomyelitis. We describe the histologic, radiologic, and diagnostic features of the tumor and outline treatment and prognosis. To our knowledge, this is the first report of multifocal Ewing sarcoma involving multiple bones in the foot. The patient provided written informed consent for print and electronic publication of this case report.

Case Report

A 19-year-old man visited our clinic with the chief complaints of left foot pain and swelling. The pain started 10 months earlier and was followed by swelling. Complaints started after a minor local trauma. The man sought outside medical attention 8 months after pain onset. At his first visit at another institution, an initial radiograph was reported as normal, and all laboratory measures, including complete blood cell count (CBC) differential, ESR, and C-reactive protein (CRP) level, were within normal limits. Under the erroneous diagnosis of infection, the patient was treated with cloxacillin 500 mg 4 times a day for 4 weeks.

The patient’s pain had started 10 months before presentation (2 months after antibiotic therapy was initiated) (Figure 1). Physical examination at our institution revealed a palpable mass on the dorsum of the left foot. Anteroposterior and lateral plain radiographs showed a permeative lytic lesion with cortical destruction in the left calcaneus, navicular, cuboid, and cuneiform bones and in all metatarsal bones except the first (Figure 2). A soft-tissue mass around the involved bones was noted as well. The talus was not involved (Figure 3).

CT showed permeative destruction of left foot bones, including the calcaneus, navicular, cuboid, and cuneiform bones and all metatarsal bones except the first. Invasion through the overlying cortex of the involved bones indicated aggressive biological activity of the tumor (Figure 4). MRI showed a destructive bony lesion of the mentioned bones associated with the soft-tissue mass (Figure 3).

Bone scan showed increased uptake in the involved areas (Figure 5). Chest plain radiographs and CT showed no distant metastasis. An incisional biopsy was performed, and histopathology showed a malignant small round cell tumor, identified as Ewing sarcoma (Figure 6). An immunohistochemistry study demonstrated positive CD99 and negative cytokeratin, leukocyte common antigen, desmin, and synaptophysin.

The patient was started on 4 cycles of adjuvant chemotherapy. Cycles 1 and 3 involved cyclophosphamide 2 g, vincristine 2 g, and doxorubicin 50 mg; cycles 2 and 4 involved ifosfamide 3.5 g and etoposide 200 mg. Tumor shrinkage occurred after chemotherapy. Clinical response to preoperative chemotherapy was documented by a decrease in tumor size at follow-ups. The patient underwent below-knee amputation.

Postoperative histopathology confirmed the diagnosis of Ewing sarcoma of the calcaneus, navicular, cuboid, and cuneiform bones and all metatarsal bones except the first (Figure 7). At 2-year follow-up, the patient had no evidence of local recurrence or distant metastasis based on chest CT and clinical examination of the affected site.

Discussion

Ewing sarcoma is the prototype of round small cell malignancies that arise from the long bones and the flat bones. It seldom involves the hands or feet. To our knowledge, this is the first report of Ewing sarcoma of the foot with multiple-bone involvement. Our literature review found a case of Ewing sarcoma of the first phalanx of the third toe, the second metatarsal bone, the cuneiform, the cuboid, and the talus, with lesser soft-tissue extension compared with our patient’s case.⁹

As this foot tumor is rare, there are few reports on its clinical aspects, appropriate treatment, and long-term outcome. For treatment of nonmetastatic Ewing sarcoma, limb-salvage surgery is advised if response has been good and there is a reasonable expectation of negative margins and good functional results.

Radiation and surgery may be part of the overall treatment plan. Radiation without surgery has a unique role in pelvic Ewing sarcoma, in contrast to extremity Ewing sarcoma. In our opinion, margins and histologic necrosis in the resection specimen are examined after surgery, and, if the margins are widely negative and histologic response is good, no further local control is advised. If the margin is positive, postoperative radiation therapy is recommended.¹ Amputation has gradually become a (rare) choice in the treatment of extremity sarcomas.⁹ In our patient’s case, surgery was preferred over radiation therapy after chemotherapy because of the low risk of local side effects and the expected high efficacy. In addition, radiation at such high doses for Ewing sarcoma in the foot causes functional impairment. Because of the multiple-bone involvement, a salvage procedure was not possible for our patient. Given the calcaneal involvement, however, below-knee amputation was considered safer than ankle disarticulation.

Multiple-bone involvement occurs in the advanced stage of Ewing sarcoma, usually after visceral and pulmonary metastases are detected.⁹ The case reported by Rammal and colleagues⁹ had both multiple-bone involvement in the foot and pulmonary metastasis. The authors indicated that hematogenous spread of the tumor was discerned because the lesions were noncontiguous.⁹ Our patient had no distant metastases. We think his tumor originated in a tarsal or midtarsal bone and extended to adjacent bones. Therefore, it probably spread through its capsular and ligamentous attachment among tarsal and midtarsal bones, as the involvement was contiguous rather than distinct.

Average delay from symptom onset to diagnosis was reported to be 34 weeks.³ Average physician delay from initial visit to correct diagnosis was reported to be 19 weeks.³ Patients may have erythema, fever, and swelling, suggestive of osteomyelitis.³ Laboratory results may show increased white blood cell count and elevated ESR and CRP level.³ In addition, needle biopsy of the tumor may reveal an appearance grossly similar to that of pus.³ Therefore, physicians may send all the tissue out for microbiological analysis (according to the erroneous diagnosis of infection) and none out for pathologic analysis. The situation can be further complicated when Ewing sarcoma occurs in the foot, an uncommon site. In this special case, multiple-bone involvement can present a misleading clinical picture of infection.¹⁰ In other words, infection is one of the best choices in the differential diagnosis.⁷ Also to be considered are multicentric giant cell tumor, fibrosarcoma,¹¹ and osteosarcoma.¹²

Ewing sarcomas are characterized as primitive malignant round cell tumors.¹ These tumors are diagnosed by neuroectodermal differentiation and by their common histologic and immunohistochemical properties.² Ewing sarcoma is the second most common malignant bone tumor in adolescents and young adults. It is the fourth most common primary malignant tumor, accounting for about 9% of all malignant tumors of bone. The most common primary bone tumors are multiple myeloma, osteosarcoma, and chondrosarcoma.³

The diaphyses of long bones (eg, femur, tibia, humerus) and flat bones (eg, pelvis, scapula) are the most commonly involved sites. Involvement of bones in the hands and feet is uncommon (3%-5% of reported cases).⁴ The foot bones most commonly involved include the calcaneus and the metatarsals, in the series by Casadei and colleagues.⁵

About 90% of Ewing sarcoma cases present before age 20 years (mean age, 13 years).⁶ Typical presentation is that of localized pain at the involved site. Some patients have systemic symptoms, such as fever, malaise, weight loss, leukocytosis, and increased erythrocyte sedimentation rate (ESR) mimicking infection. Radiographically, Ewing sarcoma appears as a permeative destructive bone lesion with a moth-eaten appearance (almost 76% of cases).⁷ This is usually associated with lamellated periosteal new bone formation or an “onion skin” appearance. Less commonly, a sunburst configuration with an associated soft-tissue mass can be seen. Computed tomography (CT) and magnetic resonance imaging (MRI) show the osseous extent of the tumor and the presence or absence of the soft-tissue component of the tumor. Radionuclide bone scans show increased technetium-99m methylene diphosphonate accumulation and are typically hot.⁶

Histopathologically, the tumor is composed of small, uniformly sized cells characterized by an almost clear eosinophilic cytoplasm and very little intercellular matrix. There are lobules and strands divided by prominent septa. Macroscopically, appearance can range from a soft, fleshy solid mass to an almost liquid form, as the lesion does not produce any matrix. At time of surgery, the tumor may have a liquefied component and the appearance of pus.⁶ Prognostic factors are tumor site in foot and treatment according to the series by Casadei and colleagues.⁵ Patients with large central tumors, especially in the pelvis, have worse outcomes than patients with distal tumors.⁸

In this article, we report a case of multifocal Ewing sarcoma involving multiple bones in the foot. Given the multifocal nature of the disease confined to the foot, the initial impression was that of osteomyelitis. We describe the histologic, radiologic, and diagnostic features of the tumor and outline treatment and prognosis. To our knowledge, this is the first report of multifocal Ewing sarcoma involving multiple bones in the foot. The patient provided written informed consent for print and electronic publication of this case report.

Case Report

A 19-year-old man visited our clinic with the chief complaints of left foot pain and swelling. The pain started 10 months earlier and was followed by swelling. Complaints started after a minor local trauma. The man sought outside medical attention 8 months after pain onset. At his first visit at another institution, an initial radiograph was reported as normal, and all laboratory measures, including complete blood cell count (CBC) differential, ESR, and C-reactive protein (CRP) level, were within normal limits. Under the erroneous diagnosis of infection, the patient was treated with cloxacillin 500 mg 4 times a day for 4 weeks.

The patient’s pain had started 10 months before presentation (2 months after antibiotic therapy was initiated) (Figure 1). Physical examination at our institution revealed a palpable mass on the dorsum of the left foot. Anteroposterior and lateral plain radiographs showed a permeative lytic lesion with cortical destruction in the left calcaneus, navicular, cuboid, and cuneiform bones and in all metatarsal bones except the first (Figure 2). A soft-tissue mass around the involved bones was noted as well. The talus was not involved (Figure 3).

CT showed permeative destruction of left foot bones, including the calcaneus, navicular, cuboid, and cuneiform bones and all metatarsal bones except the first. Invasion through the overlying cortex of the involved bones indicated aggressive biological activity of the tumor (Figure 4). MRI showed a destructive bony lesion of the mentioned bones associated with the soft-tissue mass (Figure 3).

Bone scan showed increased uptake in the involved areas (Figure 5). Chest plain radiographs and CT showed no distant metastasis. An incisional biopsy was performed, and histopathology showed a malignant small round cell tumor, identified as Ewing sarcoma (Figure 6). An immunohistochemistry study demonstrated positive CD99 and negative cytokeratin, leukocyte common antigen, desmin, and synaptophysin.

The patient was started on 4 cycles of adjuvant chemotherapy. Cycles 1 and 3 involved cyclophosphamide 2 g, vincristine 2 g, and doxorubicin 50 mg; cycles 2 and 4 involved ifosfamide 3.5 g and etoposide 200 mg. Tumor shrinkage occurred after chemotherapy. Clinical response to preoperative chemotherapy was documented by a decrease in tumor size at follow-ups. The patient underwent below-knee amputation.

Postoperative histopathology confirmed the diagnosis of Ewing sarcoma of the calcaneus, navicular, cuboid, and cuneiform bones and all metatarsal bones except the first (Figure 7). At 2-year follow-up, the patient had no evidence of local recurrence or distant metastasis based on chest CT and clinical examination of the affected site.

Discussion

Ewing sarcoma is the prototype of round small cell malignancies that arise from the long bones and the flat bones. It seldom involves the hands or feet. To our knowledge, this is the first report of Ewing sarcoma of the foot with multiple-bone involvement. Our literature review found a case of Ewing sarcoma of the first phalanx of the third toe, the second metatarsal bone, the cuneiform, the cuboid, and the talus, with lesser soft-tissue extension compared with our patient’s case.⁹

As this foot tumor is rare, there are few reports on its clinical aspects, appropriate treatment, and long-term outcome. For treatment of nonmetastatic Ewing sarcoma, limb-salvage surgery is advised if response has been good and there is a reasonable expectation of negative margins and good functional results.

Radiation and surgery may be part of the overall treatment plan. Radiation without surgery has a unique role in pelvic Ewing sarcoma, in contrast to extremity Ewing sarcoma. In our opinion, margins and histologic necrosis in the resection specimen are examined after surgery, and, if the margins are widely negative and histologic response is good, no further local control is advised. If the margin is positive, postoperative radiation therapy is recommended.¹ Amputation has gradually become a (rare) choice in the treatment of extremity sarcomas.⁹ In our patient’s case, surgery was preferred over radiation therapy after chemotherapy because of the low risk of local side effects and the expected high efficacy. In addition, radiation at such high doses for Ewing sarcoma in the foot causes functional impairment. Because of the multiple-bone involvement, a salvage procedure was not possible for our patient. Given the calcaneal involvement, however, below-knee amputation was considered safer than ankle disarticulation.

Multiple-bone involvement occurs in the advanced stage of Ewing sarcoma, usually after visceral and pulmonary metastases are detected.⁹ The case reported by Rammal and colleagues⁹ had both multiple-bone involvement in the foot and pulmonary metastasis. The authors indicated that hematogenous spread of the tumor was discerned because the lesions were noncontiguous.⁹ Our patient had no distant metastases. We think his tumor originated in a tarsal or midtarsal bone and extended to adjacent bones. Therefore, it probably spread through its capsular and ligamentous attachment among tarsal and midtarsal bones, as the involvement was contiguous rather than distinct.

Average delay from symptom onset to diagnosis was reported to be 34 weeks.³ Average physician delay from initial visit to correct diagnosis was reported to be 19 weeks.³ Patients may have erythema, fever, and swelling, suggestive of osteomyelitis.³ Laboratory results may show increased white blood cell count and elevated ESR and CRP level.³ In addition, needle biopsy of the tumor may reveal an appearance grossly similar to that of pus.³ Therefore, physicians may send all the tissue out for microbiological analysis (according to the erroneous diagnosis of infection) and none out for pathologic analysis. The situation can be further complicated when Ewing sarcoma occurs in the foot, an uncommon site. In this special case, multiple-bone involvement can present a misleading clinical picture of infection.¹⁰ In other words, infection is one of the best choices in the differential diagnosis.⁷ Also to be considered are multicentric giant cell tumor, fibrosarcoma,¹¹ and osteosarcoma.¹²

Ewing sarcomas are characterized as primitive malignant round cell tumors.¹ These tumors are diagnosed by neuroectodermal differentiation and by their common histologic and immunohistochemical properties.² Ewing sarcoma is the second most common malignant bone tumor in adolescents and young adults. It is the fourth most common primary malignant tumor, accounting for about 9% of all malignant tumors of bone. The most common primary bone tumors are multiple myeloma, osteosarcoma, and chondrosarcoma.³

The diaphyses of long bones (eg, femur, tibia, humerus) and flat bones (eg, pelvis, scapula) are the most commonly involved sites. Involvement of bones in the hands and feet is uncommon (3%-5% of reported cases).⁴ The foot bones most commonly involved include the calcaneus and the metatarsals, in the series by Casadei and colleagues.⁵

About 90% of Ewing sarcoma cases present before age 20 years (mean age, 13 years).⁶ Typical presentation is that of localized pain at the involved site. Some patients have systemic symptoms, such as fever, malaise, weight loss, leukocytosis, and increased erythrocyte sedimentation rate (ESR) mimicking infection. Radiographically, Ewing sarcoma appears as a permeative destructive bone lesion with a moth-eaten appearance (almost 76% of cases).⁷ This is usually associated with lamellated periosteal new bone formation or an “onion skin” appearance. Less commonly, a sunburst configuration with an associated soft-tissue mass can be seen. Computed tomography (CT) and magnetic resonance imaging (MRI) show the osseous extent of the tumor and the presence or absence of the soft-tissue component of the tumor. Radionuclide bone scans show increased technetium-99m methylene diphosphonate accumulation and are typically hot.⁶

Histopathologically, the tumor is composed of small, uniformly sized cells characterized by an almost clear eosinophilic cytoplasm and very little intercellular matrix. There are lobules and strands divided by prominent septa. Macroscopically, appearance can range from a soft, fleshy solid mass to an almost liquid form, as the lesion does not produce any matrix. At time of surgery, the tumor may have a liquefied component and the appearance of pus.⁶ Prognostic factors are tumor site in foot and treatment according to the series by Casadei and colleagues.⁵ Patients with large central tumors, especially in the pelvis, have worse outcomes than patients with distal tumors.⁸

In this article, we report a case of multifocal Ewing sarcoma involving multiple bones in the foot. Given the multifocal nature of the disease confined to the foot, the initial impression was that of osteomyelitis. We describe the histologic, radiologic, and diagnostic features of the tumor and outline treatment and prognosis. To our knowledge, this is the first report of multifocal Ewing sarcoma involving multiple bones in the foot. The patient provided written informed consent for print and electronic publication of this case report.

Case Report

A 19-year-old man visited our clinic with the chief complaints of left foot pain and swelling. The pain started 10 months earlier and was followed by swelling. Complaints started after a minor local trauma. The man sought outside medical attention 8 months after pain onset. At his first visit at another institution, an initial radiograph was reported as normal, and all laboratory measures, including complete blood cell count (CBC) differential, ESR, and C-reactive protein (CRP) level, were within normal limits. Under the erroneous diagnosis of infection, the patient was treated with cloxacillin 500 mg 4 times a day for 4 weeks.

The patient’s pain had started 10 months before presentation (2 months after antibiotic therapy was initiated) (Figure 1). Physical examination at our institution revealed a palpable mass on the dorsum of the left foot. Anteroposterior and lateral plain radiographs showed a permeative lytic lesion with cortical destruction in the left calcaneus, navicular, cuboid, and cuneiform bones and in all metatarsal bones except the first (Figure 2). A soft-tissue mass around the involved bones was noted as well. The talus was not involved (Figure 3).

CT showed permeative destruction of left foot bones, including the calcaneus, navicular, cuboid, and cuneiform bones and all metatarsal bones except the first. Invasion through the overlying cortex of the involved bones indicated aggressive biological activity of the tumor (Figure 4). MRI showed a destructive bony lesion of the mentioned bones associated with the soft-tissue mass (Figure 3).

Bone scan showed increased uptake in the involved areas (Figure 5). Chest plain radiographs and CT showed no distant metastasis. An incisional biopsy was performed, and histopathology showed a malignant small round cell tumor, identified as Ewing sarcoma (Figure 6). An immunohistochemistry study demonstrated positive CD99 and negative cytokeratin, leukocyte common antigen, desmin, and synaptophysin.

The patient was started on 4 cycles of adjuvant chemotherapy. Cycles 1 and 3 involved cyclophosphamide 2 g, vincristine 2 g, and doxorubicin 50 mg; cycles 2 and 4 involved ifosfamide 3.5 g and etoposide 200 mg. Tumor shrinkage occurred after chemotherapy. Clinical response to preoperative chemotherapy was documented by a decrease in tumor size at follow-ups. The patient underwent below-knee amputation.

Postoperative histopathology confirmed the diagnosis of Ewing sarcoma of the calcaneus, navicular, cuboid, and cuneiform bones and all metatarsal bones except the first (Figure 7). At 2-year follow-up, the patient had no evidence of local recurrence or distant metastasis based on chest CT and clinical examination of the affected site.

Discussion

Ewing sarcoma is the prototype of round small cell malignancies that arise from the long bones and the flat bones. It seldom involves the hands or feet. To our knowledge, this is the first report of Ewing sarcoma of the foot with multiple-bone involvement. Our literature review found a case of Ewing sarcoma of the first phalanx of the third toe, the second metatarsal bone, the cuneiform, the cuboid, and the talus, with lesser soft-tissue extension compared with our patient’s case.⁹

As this foot tumor is rare, there are few reports on its clinical aspects, appropriate treatment, and long-term outcome. For treatment of nonmetastatic Ewing sarcoma, limb-salvage surgery is advised if response has been good and there is a reasonable expectation of negative margins and good functional results.

Radiation and surgery may be part of the overall treatment plan. Radiation without surgery has a unique role in pelvic Ewing sarcoma, in contrast to extremity Ewing sarcoma. In our opinion, margins and histologic necrosis in the resection specimen are examined after surgery, and, if the margins are widely negative and histologic response is good, no further local control is advised. If the margin is positive, postoperative radiation therapy is recommended.¹ Amputation has gradually become a (rare) choice in the treatment of extremity sarcomas.⁹ In our patient’s case, surgery was preferred over radiation therapy after chemotherapy because of the low risk of local side effects and the expected high efficacy. In addition, radiation at such high doses for Ewing sarcoma in the foot causes functional impairment. Because of the multiple-bone involvement, a salvage procedure was not possible for our patient. Given the calcaneal involvement, however, below-knee amputation was considered safer than ankle disarticulation.

Multiple-bone involvement occurs in the advanced stage of Ewing sarcoma, usually after visceral and pulmonary metastases are detected.⁹ The case reported by Rammal and colleagues⁹ had both multiple-bone involvement in the foot and pulmonary metastasis. The authors indicated that hematogenous spread of the tumor was discerned because the lesions were noncontiguous.⁹ Our patient had no distant metastases. We think his tumor originated in a tarsal or midtarsal bone and extended to adjacent bones. Therefore, it probably spread through its capsular and ligamentous attachment among tarsal and midtarsal bones, as the involvement was contiguous rather than distinct.

Average delay from symptom onset to diagnosis was reported to be 34 weeks.³ Average physician delay from initial visit to correct diagnosis was reported to be 19 weeks.³ Patients may have erythema, fever, and swelling, suggestive of osteomyelitis.³ Laboratory results may show increased white blood cell count and elevated ESR and CRP level.³ In addition, needle biopsy of the tumor may reveal an appearance grossly similar to that of pus.³ Therefore, physicians may send all the tissue out for microbiological analysis (according to the erroneous diagnosis of infection) and none out for pathologic analysis. The situation can be further complicated when Ewing sarcoma occurs in the foot, an uncommon site. In this special case, multiple-bone involvement can present a misleading clinical picture of infection.¹⁰ In other words, infection is one of the best choices in the differential diagnosis.⁷ Also to be considered are multicentric giant cell tumor, fibrosarcoma,¹¹ and osteosarcoma.¹²

Children often present to orthopedic surgeons with angular deformities about the knee. Temporary hemiepiphysiodesis, which is a frequently performed procedure to address such deformities, is safe and reversible. Specifically, tension band plating has become one of the most commonly performed techniques, especially given its low complication rates and minimally invasive nature.^1-4 Complications reported with this method include mechanical hardware failure,⁵ implant migration,⁴ and recurvatum.³

We present an unreported complication of a synovial fistula formation after the removal of a tension band plate in a child who had achieved appropriate correction of her genu valgum. The patient and her family provided written informed consent for print and electronic publication of this case report.

Case Report

An 11-year-old girl presented to the pediatric orthopedics clinic with concern for genu valgum of the right lower extremity. She underwent a right proximal tibia medial hemiepiphysiodesis via tension band plating technique. Her clinic visit 4 weeks after surgery showed well-healed incisions and no signs of infection. She achieved appropriate correction and underwent hardware removal approximately 6 months after her initial surgery. 

One month after hardware removal, the patient began to notice increased swelling and erythema around her incision site with associated pain. No fluid or drainage was seen at that time. She underwent irrigation and débridement shortly thereafter, and the wound was left open for wet-to-dry dressing changes (Figure 1). Intraoperative cultures were negative, but the patient received empiric antibiotic therapy. She continued to have difficulty with wound healing for the next month and was referred to plastic surgery. She underwent repeat irrigation and débridement, followed by coverage with a split-thickness skin graft by the plastic surgery service. Intraoperative cultures were again negative. During both irrigation and débridement procedures, care was taken to remain superficial and not violate the knee capsule.

At her 2-week postoperative check, the bolster covering the split thickness skin graft was removed, which revealed a 2×2-mm area of clear erosion near the central portion of her wound with synovial fluid drainage (Figure 2). Because of concern for a synovial fistula, magnetic resonance imaging (MRI) of the right knee was obtained, which confirmed the synovial fistula (Figures 3A, 3B). The coronal cut on MRI clearly showed the fistula with synovial fluid tracking into the epiphyseal screw tract through the breached capsule and to the level of the skin. She was immobilized in a long leg cast with the knee in extension for 6 weeks. Upon return, her fistula had closed, and she has not had any more wound issues. 

Discussion

To our knowledge, this is the first report of a synovial fistula after temporary hemiepiphysiodesis performed via tension band plating. Capsular knee anatomy may explain the etiology of the synovial fistula after hardware removal. The medial knee capsule composition and attachment sites have been extensively studied.⁶ In contrast to other joints, such as the shoulder, elbow, ankle, and hip, the metaphysis of the knee lies outside the capsule because the capsule inserts proximal at the level of the physis.⁷ During tension band plating, the epiphyseal screw breaches the capsule but serves as a plug while in place, which prevents the formation of a synovial fistula. When the screw is removed, the capsular rent spontaneously closes in almost all cases. However, the opportunity exists for a synovial fistula to form while the capsule heals, as evidenced by the current case. Such an issue does not apply to the metaphyseal screw because it is inserted outside the capsule.  

Although it is possible that the synovial fistula was inadvertently created during one of the irrigation and débridement procedures, this is very unlikely. The surgeons who performed these washout procedures are knowledgeable and familiar with knee anatomy. Both irrigation and débridement procedures were superficial, and care was taken not to violate the knee capsule.

A synovial fistula after knee surgery is rare. Larsen⁸ described the fistula as a phenomenon that develops when excessive synovial fluid forces its way through a synovial incision with knee flexion and muscle contraction. Such a complication is most routinely described after knee arthroscopy. Proffer and colleagues⁹ reported an incidence of 6.1 per 1000 after knee arthroscopies. The average number of days until fistula diagnosis was 6 days (range, 3-10 days). All fistulae were treated with immobilization and closed after an average of 9 days (range, 7-14 days). There were no associated infections, although prophylactic antibiotics were given. A national survey found that knee fistulae accounted for only 3.2% of all complications of knee arthroscopy.¹⁰ 

The treatment for a synovial fistula is largely nonoperative. Most will resolve with a brief period of immobilization, which allows the fistula to close.^9-10 Literature addressing fistulae that fail to heal with nonoperative treatment is limited. Excision and direct closure of the fistula, especially when chronic, often proves futile and leads to a high recurrence rate.¹¹ An alternative but more extensive treatment involves excision and coverage with a myofascial flap.¹²

Complications reported after tension band plating are uncommon. Two studies reported no complications regarding the use of the tension band plate.^1-2 Burghardt and colleagues,⁵ in reporting the results of a multicenter survey, found that 15% of surgeons who had used tension band plating had seen a total of 65 cases of mechanical failure. In all cases, the screws, not the plate, failed. Another study reported implant migration in 1 patient but attributed the complication to a technical error from placing the distal screw too close to the physis.⁴ A third study documented that 2 patients developed clinically significant recurvatum, most likely because of anterior placement of the plate.³ It is important to identify a synovial fistula postoperatively because it provides a direct route for pathogens from the external environment to enter the intra-articular space and the opportunity for a septic joint to develop. Infection should always be ruled out and, if present, appropriately treated. 

Conclusion

Physicians performing tension band plating in the knee should be aware of the possible complication of a synovial fistula, which has traditionally been reported only in relation to knee arthroscopy. Given the proposed etiology of the synovial fistula, we recommend a brief period of immobilization of 3 to 5 days after tension band plate removal, allowing the capsular rent to heal and minimizing the risk of a synovial fistula.

Children often present to orthopedic surgeons with angular deformities about the knee. Temporary hemiepiphysiodesis, which is a frequently performed procedure to address such deformities, is safe and reversible. Specifically, tension band plating has become one of the most commonly performed techniques, especially given its low complication rates and minimally invasive nature.^1-4 Complications reported with this method include mechanical hardware failure,⁵ implant migration,⁴ and recurvatum.³

We present an unreported complication of a synovial fistula formation after the removal of a tension band plate in a child who had achieved appropriate correction of her genu valgum. The patient and her family provided written informed consent for print and electronic publication of this case report.

Case Report

An 11-year-old girl presented to the pediatric orthopedics clinic with concern for genu valgum of the right lower extremity. She underwent a right proximal tibia medial hemiepiphysiodesis via tension band plating technique. Her clinic visit 4 weeks after surgery showed well-healed incisions and no signs of infection. She achieved appropriate correction and underwent hardware removal approximately 6 months after her initial surgery. 

One month after hardware removal, the patient began to notice increased swelling and erythema around her incision site with associated pain. No fluid or drainage was seen at that time. She underwent irrigation and débridement shortly thereafter, and the wound was left open for wet-to-dry dressing changes (Figure 1). Intraoperative cultures were negative, but the patient received empiric antibiotic therapy. She continued to have difficulty with wound healing for the next month and was referred to plastic surgery. She underwent repeat irrigation and débridement, followed by coverage with a split-thickness skin graft by the plastic surgery service. Intraoperative cultures were again negative. During both irrigation and débridement procedures, care was taken to remain superficial and not violate the knee capsule.

At her 2-week postoperative check, the bolster covering the split thickness skin graft was removed, which revealed a 2×2-mm area of clear erosion near the central portion of her wound with synovial fluid drainage (Figure 2). Because of concern for a synovial fistula, magnetic resonance imaging (MRI) of the right knee was obtained, which confirmed the synovial fistula (Figures 3A, 3B). The coronal cut on MRI clearly showed the fistula with synovial fluid tracking into the epiphyseal screw tract through the breached capsule and to the level of the skin. She was immobilized in a long leg cast with the knee in extension for 6 weeks. Upon return, her fistula had closed, and she has not had any more wound issues. 

Discussion

To our knowledge, this is the first report of a synovial fistula after temporary hemiepiphysiodesis performed via tension band plating. Capsular knee anatomy may explain the etiology of the synovial fistula after hardware removal. The medial knee capsule composition and attachment sites have been extensively studied.⁶ In contrast to other joints, such as the shoulder, elbow, ankle, and hip, the metaphysis of the knee lies outside the capsule because the capsule inserts proximal at the level of the physis.⁷ During tension band plating, the epiphyseal screw breaches the capsule but serves as a plug while in place, which prevents the formation of a synovial fistula. When the screw is removed, the capsular rent spontaneously closes in almost all cases. However, the opportunity exists for a synovial fistula to form while the capsule heals, as evidenced by the current case. Such an issue does not apply to the metaphyseal screw because it is inserted outside the capsule.  

Although it is possible that the synovial fistula was inadvertently created during one of the irrigation and débridement procedures, this is very unlikely. The surgeons who performed these washout procedures are knowledgeable and familiar with knee anatomy. Both irrigation and débridement procedures were superficial, and care was taken not to violate the knee capsule.

A synovial fistula after knee surgery is rare. Larsen⁸ described the fistula as a phenomenon that develops when excessive synovial fluid forces its way through a synovial incision with knee flexion and muscle contraction. Such a complication is most routinely described after knee arthroscopy. Proffer and colleagues⁹ reported an incidence of 6.1 per 1000 after knee arthroscopies. The average number of days until fistula diagnosis was 6 days (range, 3-10 days). All fistulae were treated with immobilization and closed after an average of 9 days (range, 7-14 days). There were no associated infections, although prophylactic antibiotics were given. A national survey found that knee fistulae accounted for only 3.2% of all complications of knee arthroscopy.¹⁰ 

The treatment for a synovial fistula is largely nonoperative. Most will resolve with a brief period of immobilization, which allows the fistula to close.^9-10 Literature addressing fistulae that fail to heal with nonoperative treatment is limited. Excision and direct closure of the fistula, especially when chronic, often proves futile and leads to a high recurrence rate.¹¹ An alternative but more extensive treatment involves excision and coverage with a myofascial flap.¹²

Complications reported after tension band plating are uncommon. Two studies reported no complications regarding the use of the tension band plate.^1-2 Burghardt and colleagues,⁵ in reporting the results of a multicenter survey, found that 15% of surgeons who had used tension band plating had seen a total of 65 cases of mechanical failure. In all cases, the screws, not the plate, failed. Another study reported implant migration in 1 patient but attributed the complication to a technical error from placing the distal screw too close to the physis.⁴ A third study documented that 2 patients developed clinically significant recurvatum, most likely because of anterior placement of the plate.³ It is important to identify a synovial fistula postoperatively because it provides a direct route for pathogens from the external environment to enter the intra-articular space and the opportunity for a septic joint to develop. Infection should always be ruled out and, if present, appropriately treated. 

Conclusion

Physicians performing tension band plating in the knee should be aware of the possible complication of a synovial fistula, which has traditionally been reported only in relation to knee arthroscopy. Given the proposed etiology of the synovial fistula, we recommend a brief period of immobilization of 3 to 5 days after tension band plate removal, allowing the capsular rent to heal and minimizing the risk of a synovial fistula.

Children often present to orthopedic surgeons with angular deformities about the knee. Temporary hemiepiphysiodesis, which is a frequently performed procedure to address such deformities, is safe and reversible. Specifically, tension band plating has become one of the most commonly performed techniques, especially given its low complication rates and minimally invasive nature.^1-4 Complications reported with this method include mechanical hardware failure,⁵ implant migration,⁴ and recurvatum.³

We present an unreported complication of a synovial fistula formation after the removal of a tension band plate in a child who had achieved appropriate correction of her genu valgum. The patient and her family provided written informed consent for print and electronic publication of this case report.

Case Report

An 11-year-old girl presented to the pediatric orthopedics clinic with concern for genu valgum of the right lower extremity. She underwent a right proximal tibia medial hemiepiphysiodesis via tension band plating technique. Her clinic visit 4 weeks after surgery showed well-healed incisions and no signs of infection. She achieved appropriate correction and underwent hardware removal approximately 6 months after her initial surgery. 

One month after hardware removal, the patient began to notice increased swelling and erythema around her incision site with associated pain. No fluid or drainage was seen at that time. She underwent irrigation and débridement shortly thereafter, and the wound was left open for wet-to-dry dressing changes (Figure 1). Intraoperative cultures were negative, but the patient received empiric antibiotic therapy. She continued to have difficulty with wound healing for the next month and was referred to plastic surgery. She underwent repeat irrigation and débridement, followed by coverage with a split-thickness skin graft by the plastic surgery service. Intraoperative cultures were again negative. During both irrigation and débridement procedures, care was taken to remain superficial and not violate the knee capsule.

At her 2-week postoperative check, the bolster covering the split thickness skin graft was removed, which revealed a 2×2-mm area of clear erosion near the central portion of her wound with synovial fluid drainage (Figure 2). Because of concern for a synovial fistula, magnetic resonance imaging (MRI) of the right knee was obtained, which confirmed the synovial fistula (Figures 3A, 3B). The coronal cut on MRI clearly showed the fistula with synovial fluid tracking into the epiphyseal screw tract through the breached capsule and to the level of the skin. She was immobilized in a long leg cast with the knee in extension for 6 weeks. Upon return, her fistula had closed, and she has not had any more wound issues. 

Discussion

To our knowledge, this is the first report of a synovial fistula after temporary hemiepiphysiodesis performed via tension band plating. Capsular knee anatomy may explain the etiology of the synovial fistula after hardware removal. The medial knee capsule composition and attachment sites have been extensively studied.⁶ In contrast to other joints, such as the shoulder, elbow, ankle, and hip, the metaphysis of the knee lies outside the capsule because the capsule inserts proximal at the level of the physis.⁷ During tension band plating, the epiphyseal screw breaches the capsule but serves as a plug while in place, which prevents the formation of a synovial fistula. When the screw is removed, the capsular rent spontaneously closes in almost all cases. However, the opportunity exists for a synovial fistula to form while the capsule heals, as evidenced by the current case. Such an issue does not apply to the metaphyseal screw because it is inserted outside the capsule.  

Although it is possible that the synovial fistula was inadvertently created during one of the irrigation and débridement procedures, this is very unlikely. The surgeons who performed these washout procedures are knowledgeable and familiar with knee anatomy. Both irrigation and débridement procedures were superficial, and care was taken not to violate the knee capsule.

A synovial fistula after knee surgery is rare. Larsen⁸ described the fistula as a phenomenon that develops when excessive synovial fluid forces its way through a synovial incision with knee flexion and muscle contraction. Such a complication is most routinely described after knee arthroscopy. Proffer and colleagues⁹ reported an incidence of 6.1 per 1000 after knee arthroscopies. The average number of days until fistula diagnosis was 6 days (range, 3-10 days). All fistulae were treated with immobilization and closed after an average of 9 days (range, 7-14 days). There were no associated infections, although prophylactic antibiotics were given. A national survey found that knee fistulae accounted for only 3.2% of all complications of knee arthroscopy.¹⁰ 

The treatment for a synovial fistula is largely nonoperative. Most will resolve with a brief period of immobilization, which allows the fistula to close.^9-10 Literature addressing fistulae that fail to heal with nonoperative treatment is limited. Excision and direct closure of the fistula, especially when chronic, often proves futile and leads to a high recurrence rate.¹¹ An alternative but more extensive treatment involves excision and coverage with a myofascial flap.¹²

Complications reported after tension band plating are uncommon. Two studies reported no complications regarding the use of the tension band plate.^1-2 Burghardt and colleagues,⁵ in reporting the results of a multicenter survey, found that 15% of surgeons who had used tension band plating had seen a total of 65 cases of mechanical failure. In all cases, the screws, not the plate, failed. Another study reported implant migration in 1 patient but attributed the complication to a technical error from placing the distal screw too close to the physis.⁴ A third study documented that 2 patients developed clinically significant recurvatum, most likely because of anterior placement of the plate.³ It is important to identify a synovial fistula postoperatively because it provides a direct route for pathogens from the external environment to enter the intra-articular space and the opportunity for a septic joint to develop. Infection should always be ruled out and, if present, appropriately treated. 

Conclusion

Physicians performing tension band plating in the knee should be aware of the possible complication of a synovial fistula, which has traditionally been reported only in relation to knee arthroscopy. Given the proposed etiology of the synovial fistula, we recommend a brief period of immobilization of 3 to 5 days after tension band plate removal, allowing the capsular rent to heal and minimizing the risk of a synovial fistula.

Carcinoma of the lung is the most common lethal form of cancer in both men and women worldwide.¹ It accounts for more deaths than the next 3 most common cancers combined. In 2012, 160,000 Americans are estimated to have died from lung cancer.¹ Lung cancer is known to have a high metastatic potential for the brain, bones, adrenal glands, lungs, and liver.² Orthopedic manifestations frequently include bony metastasis, most commonly the vertebrae (42%), ribs (20%), and pelvis (18%).³ Acral metastatic disease is defined as metastasis distal to the elbow or the knee. Bony acral metastases from lung carcinoma to the upper and lower extremities are extremely uncommon, accounting for only 1% each of total bone metastases from carcinoma of the lung.³ Metastases to the bones of the hand are even rarer. Only 0.1% of metastatic disease from any type of carcinoma or sarcoma manifests as metastasis in the hand.⁴ There are only a few reports in the literature of soft-tissue or muscular metastasis to the hand from a carcinoma. Of these cases, the majority are caused by metastatic lung carcinoma.^5-9 There are no reports in the literature of metastatic disease of squamous cell origin affecting the soft tissues of the hand.

We present a case of a man with known metastatic squamous cell carcinoma of the lung who presented with acral soft-tissue metastatic disease. This report highlights a rare clinical scenario that has not been reported in the literature. The report also emphasizes a rare but important consideration for clinicians who encounter acral soft-tissue lesions in patients with a history of a primary carcinoma. The patient provided written informed consent for print and electronic publication of this case report.

Case Report

A 56-year-old man presented with right-sided pleuritic flank pain, along with a 30-lb weight loss over a 6-month period. A computed tomographic scan revealed a 5.58×3.7-cm cavitary lesion in the right lower lobe with abutment of the posterior chest wall (Figure 1). He underwent biopsy and staging, and was found to be T3N1, with biopsy-proven well-differentiated bronchogenic squamous cell carcinoma. The patient then underwent right lower and middle lobectomy with concomitant en-bloc resection of the posterior portion of ribs 7 to 11, along with mediastinal lymph-node dissection with negative margins. After surgery, he was treated with 4 cycles of adjuvant chemotherapy with cisplatin and docetaxel.

Six months after surgery, the patient began to complain of right-hand pain isolated to the thenar eminence. He also described swelling and significant pain with active or passive movement of the thumb and with relatively mild-to-moderate palpation of the area. The patient reported that the functioning of his thumb deteriorated rapidly over the course of about 1 month. On physical examination, he was neurovascularly intact with no apparent deficit in sensation of his right hand. There was no erythema or overlying skin changes. His right thenar eminence was mildly enlarged as compared with the left, and a firm, focal mass was readily palpated. Range of motion at the metacarpophalangeal joint of the thumb and index finger was limited because of pain. Thumb opposition was markedly limited. After a detailed history and physical examination, we were concerned about possible deep space infection, old hematoma, or possible metastatic disease. Magnetic resonance imaging (MRI) was ordered to evaluate the palpable mass.

Radiographically, localized soft-tissue swelling was present on the palmar surface of the hand obliquely overlying the index finger metacarpal (Figures 2, 3). On MRI, the lesion measured approximately 1.8×3.3 cm and was isointense to slightly hyperintense diffusely with central hyperintensity on T1 images (Figure 4). On T2 and short tau inversion recovery images, the lesion was more strikingly hyperintense and infiltrative in appearance (Figure 5). Postcontrast images showed avid enhancement peripherally, with central nonenhancement consistent with necrosis in the adductor pollicis.

We performed a biopsy of the lesion with the aid of immediate adequacy by fine needle aspiration cytology. We saw mitotically active malignant cells with large nuclei, high nuclear-to-cytoplasmic ratios, nucleoli, and dense cytoplasm, suggesting a metastatic squamous cell carcinoma. Because infection was part of the differential, it is pertinent to note that there was no significant inflammatory infiltrate. The core biopsy was consistent with metastatic lung cancer (Figure 6).

Discussion

This patient presented an interesting diagnostic challenge, particularly because of his previous malignancy. The differential diagnosis of acute onset thenar pain without history of trauma would include encompassing soft-tissue abscess, osteomyelitis, and infectious myositis. Soft-tissue hematoma is also in the differential for this patient, especially given the malignancy. Bony metastasis should be considered in this patient given the propensity of lung carcinoma to metastasize to bone. The location would certainly be atypical, with metastasis to the bones of the forearm or hand representing only 0.1% of all metastasis of any type of primary carcinoma or sarcoma.⁴ Primary bone or soft-tissue sarcoma should also be considered. Some authors have also suggested that necrosis, peritumoral edema-like signal, and lobulation are more common with skeletal muscle metastasis than with a primary sarcoma.¹⁰ In this case, the degree of surrounding postcontrast enhancement made simple muscle tear with hematoma unlikely, despite the  presence of increased T1 signal. The lack of evidence for localized infection and the presence of a firm focal mass on physical examination made tumor more likely than infection.        

Acrometastasis

Metastatic disease distal to the elbow and knee is very rare; specifically, metastatic disease of the hands or feet accounts for approximately 0.1% of all metastases.⁴ Carcinoma of the lung accounts for 44% to 47% of all acrometastasis.^11,12 When hand acrometastasis is considered, the right hand accounts for 55% of bony cases, likely because of hand dominance, although approximately 10% of patients had bilateral acral metastatic disease.¹² The underlying mechanism of acrometastasis remains unclear; however, some authors have postulated that it may result from an increase in vascularity or a trauma to the affected extremity.^12,13 Flynn and colleagues¹² reviewed the literature and reported a total of 257 cases of acral metastasis to the hand; they found that the median age at presentation was 58 years. Men were more than twice as likely to be affected when compared with women. Most commonly, the primary malignancies were in the lung (44%), kidney (12%), and breast (10%). The authors also reported less common cases of acral metastasis with primary malignancies located in the stomach, liver, rectum, prostate, and colon. Most commonly, these metastases were found in the distal phalynx, followed by the metacarpals, proximal phalynx, and middle phalynx.¹²

Soft-Tissue Metastasis

Skeletal muscle metastasis occurs in 0.8% to 17.5% of metastatic neoplasms.^14-17 Studies in lung cancer patients have also revealed a low prevalence of muscular metastasis (0% to 0.8%).¹⁶ The rarity of muscular metastatic disease has been attributed to local inhibition of tumor survival secondary to muscle contraction, increased diffusing capacity of enzymes and immune cells, and extreme variability in blood flow and pH, lactate, and oxygen concentration. Skeletal muscular metastases most commonly arise from the lung, kidneys, colon, or melanoma.¹⁶ In a recent large series of more than 1400 patients imaged for soft-tissue masses, 2.5% were metastatic.¹⁸ There are only 2 reports of soft-tissue metastatic disease involving the hand: one from a patient with a thyroid carcinoma and the other from a patient with a lung adenocarcinoma.¹⁸ Soft-tissue metastatic disease from squamous cell carcinoma distal to the wrist has never been reported in the literature.  

Acral Soft-Tissue Metastasis

A review from 2012 found 264 cases of skeletal muscle metastasis from 151 articles.⁶ Only 2 (0.75%) of these patients, as reported above, had a soft-tissue metastasis distal to the wrist.^6,17

Conclusion

We report the first known case of a soft-tissue metastasis distal to the wrist from a primary bronchogenic squamous cell carcinoma. This report highlights the extremely uncommon presentation of soft-tissue acral metastatic disease of a bronchogenic squamous cell carcinoma of the lung. Although exceedingly rare, oncologists and physicians who manage pathology of the hand should consider metastatic disease when evaluating a patient with complaints of hand pain and a soft-tissue mass, especially in a patient with a known primary malignancy.

Carcinoma of the lung is the most common lethal form of cancer in both men and women worldwide.¹ It accounts for more deaths than the next 3 most common cancers combined. In 2012, 160,000 Americans are estimated to have died from lung cancer.¹ Lung cancer is known to have a high metastatic potential for the brain, bones, adrenal glands, lungs, and liver.² Orthopedic manifestations frequently include bony metastasis, most commonly the vertebrae (42%), ribs (20%), and pelvis (18%).³ Acral metastatic disease is defined as metastasis distal to the elbow or the knee. Bony acral metastases from lung carcinoma to the upper and lower extremities are extremely uncommon, accounting for only 1% each of total bone metastases from carcinoma of the lung.³ Metastases to the bones of the hand are even rarer. Only 0.1% of metastatic disease from any type of carcinoma or sarcoma manifests as metastasis in the hand.⁴ There are only a few reports in the literature of soft-tissue or muscular metastasis to the hand from a carcinoma. Of these cases, the majority are caused by metastatic lung carcinoma.^5-9 There are no reports in the literature of metastatic disease of squamous cell origin affecting the soft tissues of the hand.

We present a case of a man with known metastatic squamous cell carcinoma of the lung who presented with acral soft-tissue metastatic disease. This report highlights a rare clinical scenario that has not been reported in the literature. The report also emphasizes a rare but important consideration for clinicians who encounter acral soft-tissue lesions in patients with a history of a primary carcinoma. The patient provided written informed consent for print and electronic publication of this case report.

Case Report

A 56-year-old man presented with right-sided pleuritic flank pain, along with a 30-lb weight loss over a 6-month period. A computed tomographic scan revealed a 5.58×3.7-cm cavitary lesion in the right lower lobe with abutment of the posterior chest wall (Figure 1). He underwent biopsy and staging, and was found to be T3N1, with biopsy-proven well-differentiated bronchogenic squamous cell carcinoma. The patient then underwent right lower and middle lobectomy with concomitant en-bloc resection of the posterior portion of ribs 7 to 11, along with mediastinal lymph-node dissection with negative margins. After surgery, he was treated with 4 cycles of adjuvant chemotherapy with cisplatin and docetaxel.

Six months after surgery, the patient began to complain of right-hand pain isolated to the thenar eminence. He also described swelling and significant pain with active or passive movement of the thumb and with relatively mild-to-moderate palpation of the area. The patient reported that the functioning of his thumb deteriorated rapidly over the course of about 1 month. On physical examination, he was neurovascularly intact with no apparent deficit in sensation of his right hand. There was no erythema or overlying skin changes. His right thenar eminence was mildly enlarged as compared with the left, and a firm, focal mass was readily palpated. Range of motion at the metacarpophalangeal joint of the thumb and index finger was limited because of pain. Thumb opposition was markedly limited. After a detailed history and physical examination, we were concerned about possible deep space infection, old hematoma, or possible metastatic disease. Magnetic resonance imaging (MRI) was ordered to evaluate the palpable mass.

Radiographically, localized soft-tissue swelling was present on the palmar surface of the hand obliquely overlying the index finger metacarpal (Figures 2, 3). On MRI, the lesion measured approximately 1.8×3.3 cm and was isointense to slightly hyperintense diffusely with central hyperintensity on T1 images (Figure 4). On T2 and short tau inversion recovery images, the lesion was more strikingly hyperintense and infiltrative in appearance (Figure 5). Postcontrast images showed avid enhancement peripherally, with central nonenhancement consistent with necrosis in the adductor pollicis.

We performed a biopsy of the lesion with the aid of immediate adequacy by fine needle aspiration cytology. We saw mitotically active malignant cells with large nuclei, high nuclear-to-cytoplasmic ratios, nucleoli, and dense cytoplasm, suggesting a metastatic squamous cell carcinoma. Because infection was part of the differential, it is pertinent to note that there was no significant inflammatory infiltrate. The core biopsy was consistent with metastatic lung cancer (Figure 6).

Discussion

This patient presented an interesting diagnostic challenge, particularly because of his previous malignancy. The differential diagnosis of acute onset thenar pain without history of trauma would include encompassing soft-tissue abscess, osteomyelitis, and infectious myositis. Soft-tissue hematoma is also in the differential for this patient, especially given the malignancy. Bony metastasis should be considered in this patient given the propensity of lung carcinoma to metastasize to bone. The location would certainly be atypical, with metastasis to the bones of the forearm or hand representing only 0.1% of all metastasis of any type of primary carcinoma or sarcoma.⁴ Primary bone or soft-tissue sarcoma should also be considered. Some authors have also suggested that necrosis, peritumoral edema-like signal, and lobulation are more common with skeletal muscle metastasis than with a primary sarcoma.¹⁰ In this case, the degree of surrounding postcontrast enhancement made simple muscle tear with hematoma unlikely, despite the  presence of increased T1 signal. The lack of evidence for localized infection and the presence of a firm focal mass on physical examination made tumor more likely than infection.        

Acrometastasis

Metastatic disease distal to the elbow and knee is very rare; specifically, metastatic disease of the hands or feet accounts for approximately 0.1% of all metastases.⁴ Carcinoma of the lung accounts for 44% to 47% of all acrometastasis.^11,12 When hand acrometastasis is considered, the right hand accounts for 55% of bony cases, likely because of hand dominance, although approximately 10% of patients had bilateral acral metastatic disease.¹² The underlying mechanism of acrometastasis remains unclear; however, some authors have postulated that it may result from an increase in vascularity or a trauma to the affected extremity.^12,13 Flynn and colleagues¹² reviewed the literature and reported a total of 257 cases of acral metastasis to the hand; they found that the median age at presentation was 58 years. Men were more than twice as likely to be affected when compared with women. Most commonly, the primary malignancies were in the lung (44%), kidney (12%), and breast (10%). The authors also reported less common cases of acral metastasis with primary malignancies located in the stomach, liver, rectum, prostate, and colon. Most commonly, these metastases were found in the distal phalynx, followed by the metacarpals, proximal phalynx, and middle phalynx.¹²

Soft-Tissue Metastasis

Skeletal muscle metastasis occurs in 0.8% to 17.5% of metastatic neoplasms.^14-17 Studies in lung cancer patients have also revealed a low prevalence of muscular metastasis (0% to 0.8%).¹⁶ The rarity of muscular metastatic disease has been attributed to local inhibition of tumor survival secondary to muscle contraction, increased diffusing capacity of enzymes and immune cells, and extreme variability in blood flow and pH, lactate, and oxygen concentration. Skeletal muscular metastases most commonly arise from the lung, kidneys, colon, or melanoma.¹⁶ In a recent large series of more than 1400 patients imaged for soft-tissue masses, 2.5% were metastatic.¹⁸ There are only 2 reports of soft-tissue metastatic disease involving the hand: one from a patient with a thyroid carcinoma and the other from a patient with a lung adenocarcinoma.¹⁸ Soft-tissue metastatic disease from squamous cell carcinoma distal to the wrist has never been reported in the literature.  

Acral Soft-Tissue Metastasis

A review from 2012 found 264 cases of skeletal muscle metastasis from 151 articles.⁶ Only 2 (0.75%) of these patients, as reported above, had a soft-tissue metastasis distal to the wrist.^6,17

Conclusion

We report the first known case of a soft-tissue metastasis distal to the wrist from a primary bronchogenic squamous cell carcinoma. This report highlights the extremely uncommon presentation of soft-tissue acral metastatic disease of a bronchogenic squamous cell carcinoma of the lung. Although exceedingly rare, oncologists and physicians who manage pathology of the hand should consider metastatic disease when evaluating a patient with complaints of hand pain and a soft-tissue mass, especially in a patient with a known primary malignancy.

Carcinoma of the lung is the most common lethal form of cancer in both men and women worldwide.¹ It accounts for more deaths than the next 3 most common cancers combined. In 2012, 160,000 Americans are estimated to have died from lung cancer.¹ Lung cancer is known to have a high metastatic potential for the brain, bones, adrenal glands, lungs, and liver.² Orthopedic manifestations frequently include bony metastasis, most commonly the vertebrae (42%), ribs (20%), and pelvis (18%).³ Acral metastatic disease is defined as metastasis distal to the elbow or the knee. Bony acral metastases from lung carcinoma to the upper and lower extremities are extremely uncommon, accounting for only 1% each of total bone metastases from carcinoma of the lung.³ Metastases to the bones of the hand are even rarer. Only 0.1% of metastatic disease from any type of carcinoma or sarcoma manifests as metastasis in the hand.⁴ There are only a few reports in the literature of soft-tissue or muscular metastasis to the hand from a carcinoma. Of these cases, the majority are caused by metastatic lung carcinoma.^5-9 There are no reports in the literature of metastatic disease of squamous cell origin affecting the soft tissues of the hand.

We present a case of a man with known metastatic squamous cell carcinoma of the lung who presented with acral soft-tissue metastatic disease. This report highlights a rare clinical scenario that has not been reported in the literature. The report also emphasizes a rare but important consideration for clinicians who encounter acral soft-tissue lesions in patients with a history of a primary carcinoma. The patient provided written informed consent for print and electronic publication of this case report.

Case Report

A 56-year-old man presented with right-sided pleuritic flank pain, along with a 30-lb weight loss over a 6-month period. A computed tomographic scan revealed a 5.58×3.7-cm cavitary lesion in the right lower lobe with abutment of the posterior chest wall (Figure 1). He underwent biopsy and staging, and was found to be T3N1, with biopsy-proven well-differentiated bronchogenic squamous cell carcinoma. The patient then underwent right lower and middle lobectomy with concomitant en-bloc resection of the posterior portion of ribs 7 to 11, along with mediastinal lymph-node dissection with negative margins. After surgery, he was treated with 4 cycles of adjuvant chemotherapy with cisplatin and docetaxel.

Six months after surgery, the patient began to complain of right-hand pain isolated to the thenar eminence. He also described swelling and significant pain with active or passive movement of the thumb and with relatively mild-to-moderate palpation of the area. The patient reported that the functioning of his thumb deteriorated rapidly over the course of about 1 month. On physical examination, he was neurovascularly intact with no apparent deficit in sensation of his right hand. There was no erythema or overlying skin changes. His right thenar eminence was mildly enlarged as compared with the left, and a firm, focal mass was readily palpated. Range of motion at the metacarpophalangeal joint of the thumb and index finger was limited because of pain. Thumb opposition was markedly limited. After a detailed history and physical examination, we were concerned about possible deep space infection, old hematoma, or possible metastatic disease. Magnetic resonance imaging (MRI) was ordered to evaluate the palpable mass.

Radiographically, localized soft-tissue swelling was present on the palmar surface of the hand obliquely overlying the index finger metacarpal (Figures 2, 3). On MRI, the lesion measured approximately 1.8×3.3 cm and was isointense to slightly hyperintense diffusely with central hyperintensity on T1 images (Figure 4). On T2 and short tau inversion recovery images, the lesion was more strikingly hyperintense and infiltrative in appearance (Figure 5). Postcontrast images showed avid enhancement peripherally, with central nonenhancement consistent with necrosis in the adductor pollicis.

We performed a biopsy of the lesion with the aid of immediate adequacy by fine needle aspiration cytology. We saw mitotically active malignant cells with large nuclei, high nuclear-to-cytoplasmic ratios, nucleoli, and dense cytoplasm, suggesting a metastatic squamous cell carcinoma. Because infection was part of the differential, it is pertinent to note that there was no significant inflammatory infiltrate. The core biopsy was consistent with metastatic lung cancer (Figure 6).

Discussion

This patient presented an interesting diagnostic challenge, particularly because of his previous malignancy. The differential diagnosis of acute onset thenar pain without history of trauma would include encompassing soft-tissue abscess, osteomyelitis, and infectious myositis. Soft-tissue hematoma is also in the differential for this patient, especially given the malignancy. Bony metastasis should be considered in this patient given the propensity of lung carcinoma to metastasize to bone. The location would certainly be atypical, with metastasis to the bones of the forearm or hand representing only 0.1% of all metastasis of any type of primary carcinoma or sarcoma.⁴ Primary bone or soft-tissue sarcoma should also be considered. Some authors have also suggested that necrosis, peritumoral edema-like signal, and lobulation are more common with skeletal muscle metastasis than with a primary sarcoma.¹⁰ In this case, the degree of surrounding postcontrast enhancement made simple muscle tear with hematoma unlikely, despite the  presence of increased T1 signal. The lack of evidence for localized infection and the presence of a firm focal mass on physical examination made tumor more likely than infection.        

Acrometastasis

Metastatic disease distal to the elbow and knee is very rare; specifically, metastatic disease of the hands or feet accounts for approximately 0.1% of all metastases.⁴ Carcinoma of the lung accounts for 44% to 47% of all acrometastasis.^11,12 When hand acrometastasis is considered, the right hand accounts for 55% of bony cases, likely because of hand dominance, although approximately 10% of patients had bilateral acral metastatic disease.¹² The underlying mechanism of acrometastasis remains unclear; however, some authors have postulated that it may result from an increase in vascularity or a trauma to the affected extremity.^12,13 Flynn and colleagues¹² reviewed the literature and reported a total of 257 cases of acral metastasis to the hand; they found that the median age at presentation was 58 years. Men were more than twice as likely to be affected when compared with women. Most commonly, the primary malignancies were in the lung (44%), kidney (12%), and breast (10%). The authors also reported less common cases of acral metastasis with primary malignancies located in the stomach, liver, rectum, prostate, and colon. Most commonly, these metastases were found in the distal phalynx, followed by the metacarpals, proximal phalynx, and middle phalynx.¹²

Soft-Tissue Metastasis

Skeletal muscle metastasis occurs in 0.8% to 17.5% of metastatic neoplasms.^14-17 Studies in lung cancer patients have also revealed a low prevalence of muscular metastasis (0% to 0.8%).¹⁶ The rarity of muscular metastatic disease has been attributed to local inhibition of tumor survival secondary to muscle contraction, increased diffusing capacity of enzymes and immune cells, and extreme variability in blood flow and pH, lactate, and oxygen concentration. Skeletal muscular metastases most commonly arise from the lung, kidneys, colon, or melanoma.¹⁶ In a recent large series of more than 1400 patients imaged for soft-tissue masses, 2.5% were metastatic.¹⁸ There are only 2 reports of soft-tissue metastatic disease involving the hand: one from a patient with a thyroid carcinoma and the other from a patient with a lung adenocarcinoma.¹⁸ Soft-tissue metastatic disease from squamous cell carcinoma distal to the wrist has never been reported in the literature.  

Acral Soft-Tissue Metastasis

A review from 2012 found 264 cases of skeletal muscle metastasis from 151 articles.⁶ Only 2 (0.75%) of these patients, as reported above, had a soft-tissue metastasis distal to the wrist.^6,17

Conclusion

We report the first known case of a soft-tissue metastasis distal to the wrist from a primary bronchogenic squamous cell carcinoma. This report highlights the extremely uncommon presentation of soft-tissue acral metastatic disease of a bronchogenic squamous cell carcinoma of the lung. Although exceedingly rare, oncologists and physicians who manage pathology of the hand should consider metastatic disease when evaluating a patient with complaints of hand pain and a soft-tissue mass, especially in a patient with a known primary malignancy.

Hip arthroscopy has experienced a dramatic increase in popularity, largely resulting from improvements in techniques and technology.^1,2 As with any procedure, there are complications associated with arthroscopy of the hip. These include neurapraxia, iatrogenic cartilage and labral injuries, postoperative bleeding, perineal skin necrosis, infection, intra-articular instrument breakage, intra-abdominal fluid extravasation, avascular necrosis, and femoral neck fracture.^1-4 Many of these have been attributed to the expected learning curve seen with any new procedure, and are less likely to occur as surgeons become more familiar with the procedure.¹ One rare but serious complication is anterior dislocation of the hip.^5-7

We present a patient who experienced an anterior hip dislocation and instability after hip arthroscopy, and was successfully treated with an anterior capsuloligamentous reconstruction. The patient provided written informed consent for print and electronic publication of this case report.

Case Report

An otherwise healthy 37-year-old woman presented to our clinic with a 6-month history of right groin pain and an occasional popping sensation during activity, which was unresponsive to hip-specific physical therapy. On physical examination, she was 5 ft 10 in tall, weighed 150 lbs, and appeared in excellent physical condition. She had no signs of systemic ligamentous laxity. She had an otherwise normal musculoskeletal, neurologic, and vascular examination in her bilateral lower extremities. She had a mild antalgic gait on the right leg.

The affected right hip could be flexed painfully to 120º, extended to 0º, adducted 20º, and abducted 45º. At 90º of flexion, her right hip could be externally rotated 30º and internally rotated 20º. Internal rotation during hip flexion beyond 90º caused sharp pain in the groin. Her normal left hip could be flexed to 120º, extended to 0º, adducted 30º, and abducted 60º. At 90º of flexion, her left hip could be externally rotated 50º and internally rotated 30º. She had negative Ober tests bilaterally but had tenderness along the right iliotibial band. She had negative Patrick and Gaenslen tests bilaterally. She had no tenderness in the area of either greater trochanter. 

Imaging evaluation included plain radiographs and a magnetic resonance arthrogram (MRA) of the right hip. The plain radiographs showed signs of femoroacetabular impingement, but no joint space narrowing, no dysplasia, and no retroversion of the acetabulum (Figures 1A, 1B). The MRA showed a degenerative peripheral tear of the anterosuperior labrum without significant cartilage wear (Figure 2).

Based upon her findings on physical examination and imaging, we recommended arthroscopic treatment of her right hip pathology. Thirteen months after initial presentation, we performed a right hip arthroscopy with the patient in the supine position. Through modified anterior and anterolateral portals, we used electrocautery to perform a capsulotomy from the 9 o’clock to 12 o’clock positions. A central compartment diagnostic arthroscopy showed mild degenerative fraying of the labrum from the 9 o’clock to 12 o’clock positions without signs of detachment. There was grade III chondral fraying near the articular margin in that same arc. The femoral articular cartilage appeared normal, as did the ligamentum teres. We used a shaver to gently débride the torn labrum down to stable tissue. The frayed cartilage on the acetabulum was also gently débrided.

Traction was released and the hip was flexed. Minimal capsular release and débridement were performed for adequate visualization of the peripheral compartment. A diagnostic examination revealed a significant cam-type impingement lesion from the 12 o’clock to 6 o’clock positions. We performed a femoral neck resection, with a proximal-distal dimension of 15 mm and a depth of 7 mm. A dynamic fluoroscopic examination of the hip joint showed no signs of impingement. In accordance with our standard protocol, the anterior capsulotomy was not repaired.

Postoperatively, the patient was instructed to perform toe-touch weight-bearing with crutches for 2 weeks and to advance to full weight-bearing over the next 2 weeks. She did not use a hip orthosis. She was also advised to avoid combined hip extension/external rotation maneuvers for the first 4 weeks. She took part in a formal hip-specific physical therapy program for a total of 12 weeks. She was seen in clinic at 2, 6, and 12 weeks postoperatively and appeared to have had a typical, uneventful course. We advised her to gradually return to normal activities as tolerated at the 12-week visit.

Four months after the procedure, the patient returned to our clinic for evaluation after a right hip dislocation. Two days prior, she was at a school function with her child and experienced sudden pain and inability to bear weight after she extended and externally rotated her right hip in a low-energy manner. She was taken to an emergency room and found to have an anterior dislocation of the right hip (Figure 3), which was concentrically reduced under anesthesia.

Upon questioning, she reported having had feelings of mild instability of the right hip during demanding activities (jogging, yoga) after sustaining a low-energy fall 1 month prior to her dislocation. On examination, she had significant apprehension about the right hip during gentle external rotation maneuvers. An MRA 2 weeks after the dislocation showed a large defect of the anterosuperior capsuloligamentous complex measuring 4 cm from medial to lateral and 2.5 cm superior to inferior (Figure 4). No loose bodies, chondral injuries, or recurrent tears of the labrum were seen. Typical postoperative changes were observed at the femoral head-neck junction.

Initially, we recommended nonoperative management with 6 weeks of toe-touch weight-bearing and strict avoidance of hip extension–external rotation maneuvers. No hip orthosis was used. After this period, the patient advanced to full weight-bearing and continued in hip-specific physical therapy. Despite continued therapy and avoidance of provocative maneuvers, the patient reported persistent feelings of right hip instability with significant apprehension during extension and external rotation of the right hip. A repeat MRA 4 months after the hip dislocation showed a persistent defect in the anterosuperior capsuloligamentous complex and no signs of avascular necrosis. After 6 months of conservative treatment, we recommended an open capsulorrhaphy of the right hip with autograft iliotibial band reconstruction of the iliofemoral ligament and capsule.

Six months after the dislocation, the patient underwent the recommended procedure. After induction of general anesthesia, she was placed in the supine position on a standard operating table. A Smith-Petersen approach was used to visualize the anterior hip structures. During deep dissection, we observed a large defect, measuring 2.5×4 cm (Figure 5A), in the anterior hip capsule, with only a thin pseudocapsule covering the femoral head. Extensive mobilization of the anterior capsule was unsuccessful.

The decision was made to harvest a graft from the patient’s ipsilateral iliotibial band. A skin incision was made over the iliotibial band in the distal midthigh region, and a 2.5×4-cm graft was harvested from the central portion of the iliotibial band. An arthrotomy was performed on the hip joint (Figure 5B). The labrum appeared healthy without recurrent tearing or fraying, and other than focal thinning on the superior acetabulum, the cartilage appeared healthy. A double-loaded anchor was placed in the supra-acetabular region, and the sutures were passed through the graft. Then, No. 2 nonabsorbable sutures were sequentially placed between the capsular remnant and the graft medially, inferiorly, and laterally. The graft was placed into position (Figure 5C) and the sutures were tied (Figure 5D).

Postoperatively, the patient was allowed toe-touch weight-bearing for 6 weeks, with strict avoidance of extension–external rotation maneuvers. She participated in a 12-week course of physical therapy with gradual advancement of activities. About a year after the capsulorrhaphy, she was able to resume all previous activities with only occasional low-level discomfort. She returned to the clinic 16 months after the capsulorrhaphy complaining of increased pain with long-distance running but denied feelings of instability. We performed an intra-articular hip injection under ultrasound guidance, which provided 100% relief of her symptoms. We obtained an MRA to evaluate for any recurrent capsular or labral injury (Figure 6). The previous anterosuperior capsular defect was not visible, and no signs of recurrent labral or cartilage injury were seen.

Discussion

With the increasing popularity of hip arthroscopy, more complications are being reported as well, including postoperative hip instability. Three separate cases of anterior hip instability have been published in the past several years.^5-7

Ranawat and colleagues⁵ were the first to report a case of postoperative anterior hip dislocation after arthroscopy. Their patient was a 52-year-old woman with right hip pain and generalized ligamentous laxity. Her preoperative radiographs showed no evidence of degenerative changes, dysplasia, or femoroacetabular impingement. An MRA showed a peripheral tear of the anterosuperior labrum. At arthroscopy, her right hip was easily distracted 2 to 3 cm with what they described as “minimal traction.” A small 1- to 2-cm capsulotomy was performed about the anterior portal. A detached labral tear was identified and repaired with an anchor, and no rim resection was performed. To improve visualization of the peripheral compartment, they extended the previous capsulotomy 1 to 2 cm and débrided the edges. A cam-type lesion was identified and resected. Lastly, they performed an anterior capsular plication, specifically including the iliofemoral ligament. Postoperatively, the patient wore a hip orthosis for 6 weeks to prevent extension and external rotation of the hip as well as a foot brace at night for 3 weeks. The patient was allowed to partially bear weight for the first 6 weeks with use of crutches. Approximately 2 months postoperatively, she slipped and fell down a short flight of stairs. She was diagnosed with an anterior hip dislocation. After successful closed reduction, she was treated conservatively with the same regimen used earlier. She remained symptomatic over the next several months with signs of instability and apprehension, and she eventually underwent a repeat hip arthroscopy. A 1- to 2-cm tear of the anterior capsule and iliofemoral ligament was treated with a revision arthroscopic capsular plication. A postoperative regimen similar to that used at the index procedure was instituted and, at most recent follow-up, she was found to have occasional pain without instability.

Matsuda⁶ reported a case of acute iatrogenic hip dislocation after arthroscopic surgery. His patient was a 39-year-old woman with a mildly retroverted acetabulum leading to impingement about the hip. She had no signs of generalized ligamentous laxity. A hip arthroscopy in the lateral position was performed, with no comment about the extent of the capsulotomy. During the procedure, about 5 mm of anterosuperior acetabulum were removed as part of arthroscopic rim trimming for treatment of the pincer lesion. A femoral osteochondroplasty was also performed (unspecified size) to restore more normal anterolateral offset. One confounding factor was that supranormal hip distraction was needed for 20 minutes to aid in removal of a metallic piece from a radiofrequency ablator, which inadvertently detached. The patient experienced an anterior hip dislocation in the recovery room and was found to be unstable during closed reduction under general anesthesia. A mini-open capsular repair was performed, which showed a 1×1.5-cm defect in the anterolateral capsule. After closure of the defect, the hip was found to be stable under fluoroscopic examination. Postoperatively, the patient was allowed to perform partial weight-bearing in a hip-knee-ankle-foot orthosis for 2 months and then a flexible hip brace for 1 month. At 15-month follow-up, her hip was stable and she was pain-free.

Benali and Katthagen⁷ highlighted the significant contribution of the labrum to hip stability in a dysplastic hip. Their patient was a 49-year-old woman with mild hip dysplasia and a degenerative bucket-handle tear of the ventrolateral labrum. The patient underwent a near-complete labral resection and rim trimming at an outside institution. The patient began full weight-bearing at 3 weeks postoperatively and noticed considerable groin and back pain (no hip orthosis use was mentioned). After failed treatment for suspected lumbar pathology, she was referred to the authors’ clinic for further evaluation. Plain radiographs showed subluxation of the left hip with degenerative changes. The patient had an uneventful left total hip arthroplasty (THA).

After reviewing the 3 reported cases of hip instability after arthroscopy, we suggest that surgeons fully recognize and appreciate the delicate balance of stability and motion provided by the static and dynamic stabilizers of the hip joint, and be cognizant of potential imbalance created by surgical intervention.^8,9 Postarthroscopic hip instability appears to be multifactorial in nature, because all of the reported cases detailed different factors, both patient- and surgeon-related, contributing to instability.

Ranawat and colleagues⁵ identified several factors that may have contributed to the anterior hip dislocation sustained by their patient, including the patient’s generalized ligamentous laxity, performance of a capsulectomy (with repair of iliofemoral ligament), and a traumatic fall. Benali and Katthagen⁷ (although they did not perform the index procedure) described the disastrous complication of overzealous labral resection and rim trimming in a patient with hip dysplasia. Matsuda⁶ performed a labral resection and rim trimming, an extended (unspecified size) capsulotomy, and also used supranormal traction for 20 minutes to remove an iatrogenic foreign body. Surgeons performing hip arthroscopies should be aware of all these factors, because many are directly controlled by the surgeon.

The only factor we feel may have contributed to hip instability in our patient was the performance of a capsulotomy without closure. Our patient was an otherwise healthy woman with no signs of ligamentous laxity, hip dysplasia, or retroversion of the acetabulum. We did not perform a labral resection or rim trimming. We use modified anterior and anterolateral portals, and electrocautery to connect the portals. This typically leads to a release of a thin strip (less than 5 mm wide) of 3 cm of capsule. Based upon findings at rare second-look arthroscopy for recurrent symptoms, Dr. Guanche has observed that the capsulotomy from the initial procedure heals with normal-appearing tissue. Also, during peripheral compartment arthroscopy, we do not routinely release the iliofemoral ligament, and the orbicular ligament is left intact. Instead, we prefer to flex the hip and débride only enough capsular tissue to allow for adequate visualization. 

Little has been published on capsulotomy closure after hip arthroscopy, and no consensus exists. Our standard practice is to not close the capsulotomy, which accords with the practice of other surgeons.⁹ There is concern, however, that extensive capsulotomy leading to injury or disruption of the iliofemoral ligament may cause anterior hip instability, driving other prominent hip arthroscopists to routinely close the capsulotomy.^9,10 Myers and colleagues¹⁰ published a recent biomechanical study on the role of the labrum and the capsular ligaments in hip stability. They concluded that the iliofemoral ligament plays a significant role in limiting external rotation and anterior translation of the femoral head, and recommended closure of the capsulotomy after arthroscopy. Of note, Dr. Guanche has performed more than 1500 hip arthroscopic procedures in the past 5 years, and we are aware of only 2 patients who have sustained anterior hip dislocations, in spite of our not closing the capsulotomy defect. This highlights an important clinical question in need of further investigation.

Our case also raises questions about the ideal postoperative regimen after standard hip arthroscopy. Although we do not routinely prescribe hip orthoses for our patients, others do.⁵ We are unaware of any proven benefit to the standard use of hip orthoses, and are concerned over the possible lack of patient compliance and of adequate restraint. We prefer to educate our patients on avoiding the “at-risk” position of hip extension and external rotation and to counsel them on gradual return to activities. Studies are needed to determine the role of these devices in hip arthroscopy, as well as the ideal postoperative activity regimen.

Our patient failed 6 months of conservative treatment after her dislocation and continued to have feelings of hip instability even during light activities. As a result of this failure and given an anatomical defect in the anterior capsuloligamentous complex, we decided our patient would be best treated with reconstruction of the defect. We did not think a revision capsular plication, as done by Ranawat and colleagues,⁵ was a reasonable option for our patient because of a large defect in the capsular tissue. Even in smaller defects, plication could potentially lead to overtightening of the capsule and subsequent overconstraint of the joint. Also, plication of defects may place excessive strain on the suture, which may fail if the repair is even mildly stressed.

Recurrent anterior hip dislocations, although rare in their own right, are much more common after THA than after hip arthroscopy.¹¹ Fujishiro and colleagues¹² described a similar technique to ours developed to treat a patient with recurrent anterior hip instability from anterior capsular insufficiency after multiple revision THA procedures. They used a Leeds-Keio artificial ligament to reconstruct the iliofemoral ligament, and this successfully treated their patient’s instability.

Conclusion

We believe this is the first report of recurrent instability after hip arthroscopy, necessitating reconstruction of the anterior capsuloligamentous complex. This case shows that reconstruction of the iliofemoral ligament with iliotibial band autograft is safe, restores hip stability without compromising function, and should be considered by any hip arthroscopist encountering a similar scenario. It also highlights the importance of the capsuloligamentous complex surrounding the hip joint for its stability and the need for further research to better delineate the indications for capsular repair/closure after capsulotomy.

Hip arthroscopy has experienced a dramatic increase in popularity, largely resulting from improvements in techniques and technology.^1,2 As with any procedure, there are complications associated with arthroscopy of the hip. These include neurapraxia, iatrogenic cartilage and labral injuries, postoperative bleeding, perineal skin necrosis, infection, intra-articular instrument breakage, intra-abdominal fluid extravasation, avascular necrosis, and femoral neck fracture.^1-4 Many of these have been attributed to the expected learning curve seen with any new procedure, and are less likely to occur as surgeons become more familiar with the procedure.¹ One rare but serious complication is anterior dislocation of the hip.^5-7

We present a patient who experienced an anterior hip dislocation and instability after hip arthroscopy, and was successfully treated with an anterior capsuloligamentous reconstruction. The patient provided written informed consent for print and electronic publication of this case report.

Case Report

An otherwise healthy 37-year-old woman presented to our clinic with a 6-month history of right groin pain and an occasional popping sensation during activity, which was unresponsive to hip-specific physical therapy. On physical examination, she was 5 ft 10 in tall, weighed 150 lbs, and appeared in excellent physical condition. She had no signs of systemic ligamentous laxity. She had an otherwise normal musculoskeletal, neurologic, and vascular examination in her bilateral lower extremities. She had a mild antalgic gait on the right leg.

The affected right hip could be flexed painfully to 120º, extended to 0º, adducted 20º, and abducted 45º. At 90º of flexion, her right hip could be externally rotated 30º and internally rotated 20º. Internal rotation during hip flexion beyond 90º caused sharp pain in the groin. Her normal left hip could be flexed to 120º, extended to 0º, adducted 30º, and abducted 60º. At 90º of flexion, her left hip could be externally rotated 50º and internally rotated 30º. She had negative Ober tests bilaterally but had tenderness along the right iliotibial band. She had negative Patrick and Gaenslen tests bilaterally. She had no tenderness in the area of either greater trochanter. 

Imaging evaluation included plain radiographs and a magnetic resonance arthrogram (MRA) of the right hip. The plain radiographs showed signs of femoroacetabular impingement, but no joint space narrowing, no dysplasia, and no retroversion of the acetabulum (Figures 1A, 1B). The MRA showed a degenerative peripheral tear of the anterosuperior labrum without significant cartilage wear (Figure 2).

Based upon her findings on physical examination and imaging, we recommended arthroscopic treatment of her right hip pathology. Thirteen months after initial presentation, we performed a right hip arthroscopy with the patient in the supine position. Through modified anterior and anterolateral portals, we used electrocautery to perform a capsulotomy from the 9 o’clock to 12 o’clock positions. A central compartment diagnostic arthroscopy showed mild degenerative fraying of the labrum from the 9 o’clock to 12 o’clock positions without signs of detachment. There was grade III chondral fraying near the articular margin in that same arc. The femoral articular cartilage appeared normal, as did the ligamentum teres. We used a shaver to gently débride the torn labrum down to stable tissue. The frayed cartilage on the acetabulum was also gently débrided.

Traction was released and the hip was flexed. Minimal capsular release and débridement were performed for adequate visualization of the peripheral compartment. A diagnostic examination revealed a significant cam-type impingement lesion from the 12 o’clock to 6 o’clock positions. We performed a femoral neck resection, with a proximal-distal dimension of 15 mm and a depth of 7 mm. A dynamic fluoroscopic examination of the hip joint showed no signs of impingement. In accordance with our standard protocol, the anterior capsulotomy was not repaired.

Postoperatively, the patient was instructed to perform toe-touch weight-bearing with crutches for 2 weeks and to advance to full weight-bearing over the next 2 weeks. She did not use a hip orthosis. She was also advised to avoid combined hip extension/external rotation maneuvers for the first 4 weeks. She took part in a formal hip-specific physical therapy program for a total of 12 weeks. She was seen in clinic at 2, 6, and 12 weeks postoperatively and appeared to have had a typical, uneventful course. We advised her to gradually return to normal activities as tolerated at the 12-week visit.

Four months after the procedure, the patient returned to our clinic for evaluation after a right hip dislocation. Two days prior, she was at a school function with her child and experienced sudden pain and inability to bear weight after she extended and externally rotated her right hip in a low-energy manner. She was taken to an emergency room and found to have an anterior dislocation of the right hip (Figure 3), which was concentrically reduced under anesthesia.

Upon questioning, she reported having had feelings of mild instability of the right hip during demanding activities (jogging, yoga) after sustaining a low-energy fall 1 month prior to her dislocation. On examination, she had significant apprehension about the right hip during gentle external rotation maneuvers. An MRA 2 weeks after the dislocation showed a large defect of the anterosuperior capsuloligamentous complex measuring 4 cm from medial to lateral and 2.5 cm superior to inferior (Figure 4). No loose bodies, chondral injuries, or recurrent tears of the labrum were seen. Typical postoperative changes were observed at the femoral head-neck junction.

Initially, we recommended nonoperative management with 6 weeks of toe-touch weight-bearing and strict avoidance of hip extension–external rotation maneuvers. No hip orthosis was used. After this period, the patient advanced to full weight-bearing and continued in hip-specific physical therapy. Despite continued therapy and avoidance of provocative maneuvers, the patient reported persistent feelings of right hip instability with significant apprehension during extension and external rotation of the right hip. A repeat MRA 4 months after the hip dislocation showed a persistent defect in the anterosuperior capsuloligamentous complex and no signs of avascular necrosis. After 6 months of conservative treatment, we recommended an open capsulorrhaphy of the right hip with autograft iliotibial band reconstruction of the iliofemoral ligament and capsule.

Six months after the dislocation, the patient underwent the recommended procedure. After induction of general anesthesia, she was placed in the supine position on a standard operating table. A Smith-Petersen approach was used to visualize the anterior hip structures. During deep dissection, we observed a large defect, measuring 2.5×4 cm (Figure 5A), in the anterior hip capsule, with only a thin pseudocapsule covering the femoral head. Extensive mobilization of the anterior capsule was unsuccessful.

The decision was made to harvest a graft from the patient’s ipsilateral iliotibial band. A skin incision was made over the iliotibial band in the distal midthigh region, and a 2.5×4-cm graft was harvested from the central portion of the iliotibial band. An arthrotomy was performed on the hip joint (Figure 5B). The labrum appeared healthy without recurrent tearing or fraying, and other than focal thinning on the superior acetabulum, the cartilage appeared healthy. A double-loaded anchor was placed in the supra-acetabular region, and the sutures were passed through the graft. Then, No. 2 nonabsorbable sutures were sequentially placed between the capsular remnant and the graft medially, inferiorly, and laterally. The graft was placed into position (Figure 5C) and the sutures were tied (Figure 5D).

Postoperatively, the patient was allowed toe-touch weight-bearing for 6 weeks, with strict avoidance of extension–external rotation maneuvers. She participated in a 12-week course of physical therapy with gradual advancement of activities. About a year after the capsulorrhaphy, she was able to resume all previous activities with only occasional low-level discomfort. She returned to the clinic 16 months after the capsulorrhaphy complaining of increased pain with long-distance running but denied feelings of instability. We performed an intra-articular hip injection under ultrasound guidance, which provided 100% relief of her symptoms. We obtained an MRA to evaluate for any recurrent capsular or labral injury (Figure 6). The previous anterosuperior capsular defect was not visible, and no signs of recurrent labral or cartilage injury were seen.

Discussion

With the increasing popularity of hip arthroscopy, more complications are being reported as well, including postoperative hip instability. Three separate cases of anterior hip instability have been published in the past several years.^5-7

Ranawat and colleagues⁵ were the first to report a case of postoperative anterior hip dislocation after arthroscopy. Their patient was a 52-year-old woman with right hip pain and generalized ligamentous laxity. Her preoperative radiographs showed no evidence of degenerative changes, dysplasia, or femoroacetabular impingement. An MRA showed a peripheral tear of the anterosuperior labrum. At arthroscopy, her right hip was easily distracted 2 to 3 cm with what they described as “minimal traction.” A small 1- to 2-cm capsulotomy was performed about the anterior portal. A detached labral tear was identified and repaired with an anchor, and no rim resection was performed. To improve visualization of the peripheral compartment, they extended the previous capsulotomy 1 to 2 cm and débrided the edges. A cam-type lesion was identified and resected. Lastly, they performed an anterior capsular plication, specifically including the iliofemoral ligament. Postoperatively, the patient wore a hip orthosis for 6 weeks to prevent extension and external rotation of the hip as well as a foot brace at night for 3 weeks. The patient was allowed to partially bear weight for the first 6 weeks with use of crutches. Approximately 2 months postoperatively, she slipped and fell down a short flight of stairs. She was diagnosed with an anterior hip dislocation. After successful closed reduction, she was treated conservatively with the same regimen used earlier. She remained symptomatic over the next several months with signs of instability and apprehension, and she eventually underwent a repeat hip arthroscopy. A 1- to 2-cm tear of the anterior capsule and iliofemoral ligament was treated with a revision arthroscopic capsular plication. A postoperative regimen similar to that used at the index procedure was instituted and, at most recent follow-up, she was found to have occasional pain without instability.

Matsuda⁶ reported a case of acute iatrogenic hip dislocation after arthroscopic surgery. His patient was a 39-year-old woman with a mildly retroverted acetabulum leading to impingement about the hip. She had no signs of generalized ligamentous laxity. A hip arthroscopy in the lateral position was performed, with no comment about the extent of the capsulotomy. During the procedure, about 5 mm of anterosuperior acetabulum were removed as part of arthroscopic rim trimming for treatment of the pincer lesion. A femoral osteochondroplasty was also performed (unspecified size) to restore more normal anterolateral offset. One confounding factor was that supranormal hip distraction was needed for 20 minutes to aid in removal of a metallic piece from a radiofrequency ablator, which inadvertently detached. The patient experienced an anterior hip dislocation in the recovery room and was found to be unstable during closed reduction under general anesthesia. A mini-open capsular repair was performed, which showed a 1×1.5-cm defect in the anterolateral capsule. After closure of the defect, the hip was found to be stable under fluoroscopic examination. Postoperatively, the patient was allowed to perform partial weight-bearing in a hip-knee-ankle-foot orthosis for 2 months and then a flexible hip brace for 1 month. At 15-month follow-up, her hip was stable and she was pain-free.

Benali and Katthagen⁷ highlighted the significant contribution of the labrum to hip stability in a dysplastic hip. Their patient was a 49-year-old woman with mild hip dysplasia and a degenerative bucket-handle tear of the ventrolateral labrum. The patient underwent a near-complete labral resection and rim trimming at an outside institution. The patient began full weight-bearing at 3 weeks postoperatively and noticed considerable groin and back pain (no hip orthosis use was mentioned). After failed treatment for suspected lumbar pathology, she was referred to the authors’ clinic for further evaluation. Plain radiographs showed subluxation of the left hip with degenerative changes. The patient had an uneventful left total hip arthroplasty (THA).

After reviewing the 3 reported cases of hip instability after arthroscopy, we suggest that surgeons fully recognize and appreciate the delicate balance of stability and motion provided by the static and dynamic stabilizers of the hip joint, and be cognizant of potential imbalance created by surgical intervention.^8,9 Postarthroscopic hip instability appears to be multifactorial in nature, because all of the reported cases detailed different factors, both patient- and surgeon-related, contributing to instability.

Ranawat and colleagues⁵ identified several factors that may have contributed to the anterior hip dislocation sustained by their patient, including the patient’s generalized ligamentous laxity, performance of a capsulectomy (with repair of iliofemoral ligament), and a traumatic fall. Benali and Katthagen⁷ (although they did not perform the index procedure) described the disastrous complication of overzealous labral resection and rim trimming in a patient with hip dysplasia. Matsuda⁶ performed a labral resection and rim trimming, an extended (unspecified size) capsulotomy, and also used supranormal traction for 20 minutes to remove an iatrogenic foreign body. Surgeons performing hip arthroscopies should be aware of all these factors, because many are directly controlled by the surgeon.

The only factor we feel may have contributed to hip instability in our patient was the performance of a capsulotomy without closure. Our patient was an otherwise healthy woman with no signs of ligamentous laxity, hip dysplasia, or retroversion of the acetabulum. We did not perform a labral resection or rim trimming. We use modified anterior and anterolateral portals, and electrocautery to connect the portals. This typically leads to a release of a thin strip (less than 5 mm wide) of 3 cm of capsule. Based upon findings at rare second-look arthroscopy for recurrent symptoms, Dr. Guanche has observed that the capsulotomy from the initial procedure heals with normal-appearing tissue. Also, during peripheral compartment arthroscopy, we do not routinely release the iliofemoral ligament, and the orbicular ligament is left intact. Instead, we prefer to flex the hip and débride only enough capsular tissue to allow for adequate visualization. 

Little has been published on capsulotomy closure after hip arthroscopy, and no consensus exists. Our standard practice is to not close the capsulotomy, which accords with the practice of other surgeons.⁹ There is concern, however, that extensive capsulotomy leading to injury or disruption of the iliofemoral ligament may cause anterior hip instability, driving other prominent hip arthroscopists to routinely close the capsulotomy.^9,10 Myers and colleagues¹⁰ published a recent biomechanical study on the role of the labrum and the capsular ligaments in hip stability. They concluded that the iliofemoral ligament plays a significant role in limiting external rotation and anterior translation of the femoral head, and recommended closure of the capsulotomy after arthroscopy. Of note, Dr. Guanche has performed more than 1500 hip arthroscopic procedures in the past 5 years, and we are aware of only 2 patients who have sustained anterior hip dislocations, in spite of our not closing the capsulotomy defect. This highlights an important clinical question in need of further investigation.

Our case also raises questions about the ideal postoperative regimen after standard hip arthroscopy. Although we do not routinely prescribe hip orthoses for our patients, others do.⁵ We are unaware of any proven benefit to the standard use of hip orthoses, and are concerned over the possible lack of patient compliance and of adequate restraint. We prefer to educate our patients on avoiding the “at-risk” position of hip extension and external rotation and to counsel them on gradual return to activities. Studies are needed to determine the role of these devices in hip arthroscopy, as well as the ideal postoperative activity regimen.

Our patient failed 6 months of conservative treatment after her dislocation and continued to have feelings of hip instability even during light activities. As a result of this failure and given an anatomical defect in the anterior capsuloligamentous complex, we decided our patient would be best treated with reconstruction of the defect. We did not think a revision capsular plication, as done by Ranawat and colleagues,⁵ was a reasonable option for our patient because of a large defect in the capsular tissue. Even in smaller defects, plication could potentially lead to overtightening of the capsule and subsequent overconstraint of the joint. Also, plication of defects may place excessive strain on the suture, which may fail if the repair is even mildly stressed.

Recurrent anterior hip dislocations, although rare in their own right, are much more common after THA than after hip arthroscopy.¹¹ Fujishiro and colleagues¹² described a similar technique to ours developed to treat a patient with recurrent anterior hip instability from anterior capsular insufficiency after multiple revision THA procedures. They used a Leeds-Keio artificial ligament to reconstruct the iliofemoral ligament, and this successfully treated their patient’s instability.

Conclusion

We believe this is the first report of recurrent instability after hip arthroscopy, necessitating reconstruction of the anterior capsuloligamentous complex. This case shows that reconstruction of the iliofemoral ligament with iliotibial band autograft is safe, restores hip stability without compromising function, and should be considered by any hip arthroscopist encountering a similar scenario. It also highlights the importance of the capsuloligamentous complex surrounding the hip joint for its stability and the need for further research to better delineate the indications for capsular repair/closure after capsulotomy.

Hip arthroscopy has experienced a dramatic increase in popularity, largely resulting from improvements in techniques and technology.^1,2 As with any procedure, there are complications associated with arthroscopy of the hip. These include neurapraxia, iatrogenic cartilage and labral injuries, postoperative bleeding, perineal skin necrosis, infection, intra-articular instrument breakage, intra-abdominal fluid extravasation, avascular necrosis, and femoral neck fracture.^1-4 Many of these have been attributed to the expected learning curve seen with any new procedure, and are less likely to occur as surgeons become more familiar with the procedure.¹ One rare but serious complication is anterior dislocation of the hip.^5-7

We present a patient who experienced an anterior hip dislocation and instability after hip arthroscopy, and was successfully treated with an anterior capsuloligamentous reconstruction. The patient provided written informed consent for print and electronic publication of this case report.

Case Report

An otherwise healthy 37-year-old woman presented to our clinic with a 6-month history of right groin pain and an occasional popping sensation during activity, which was unresponsive to hip-specific physical therapy. On physical examination, she was 5 ft 10 in tall, weighed 150 lbs, and appeared in excellent physical condition. She had no signs of systemic ligamentous laxity. She had an otherwise normal musculoskeletal, neurologic, and vascular examination in her bilateral lower extremities. She had a mild antalgic gait on the right leg.

The affected right hip could be flexed painfully to 120º, extended to 0º, adducted 20º, and abducted 45º. At 90º of flexion, her right hip could be externally rotated 30º and internally rotated 20º. Internal rotation during hip flexion beyond 90º caused sharp pain in the groin. Her normal left hip could be flexed to 120º, extended to 0º, adducted 30º, and abducted 60º. At 90º of flexion, her left hip could be externally rotated 50º and internally rotated 30º. She had negative Ober tests bilaterally but had tenderness along the right iliotibial band. She had negative Patrick and Gaenslen tests bilaterally. She had no tenderness in the area of either greater trochanter. 

Imaging evaluation included plain radiographs and a magnetic resonance arthrogram (MRA) of the right hip. The plain radiographs showed signs of femoroacetabular impingement, but no joint space narrowing, no dysplasia, and no retroversion of the acetabulum (Figures 1A, 1B). The MRA showed a degenerative peripheral tear of the anterosuperior labrum without significant cartilage wear (Figure 2).

Based upon her findings on physical examination and imaging, we recommended arthroscopic treatment of her right hip pathology. Thirteen months after initial presentation, we performed a right hip arthroscopy with the patient in the supine position. Through modified anterior and anterolateral portals, we used electrocautery to perform a capsulotomy from the 9 o’clock to 12 o’clock positions. A central compartment diagnostic arthroscopy showed mild degenerative fraying of the labrum from the 9 o’clock to 12 o’clock positions without signs of detachment. There was grade III chondral fraying near the articular margin in that same arc. The femoral articular cartilage appeared normal, as did the ligamentum teres. We used a shaver to gently débride the torn labrum down to stable tissue. The frayed cartilage on the acetabulum was also gently débrided.

Traction was released and the hip was flexed. Minimal capsular release and débridement were performed for adequate visualization of the peripheral compartment. A diagnostic examination revealed a significant cam-type impingement lesion from the 12 o’clock to 6 o’clock positions. We performed a femoral neck resection, with a proximal-distal dimension of 15 mm and a depth of 7 mm. A dynamic fluoroscopic examination of the hip joint showed no signs of impingement. In accordance with our standard protocol, the anterior capsulotomy was not repaired.

Postoperatively, the patient was instructed to perform toe-touch weight-bearing with crutches for 2 weeks and to advance to full weight-bearing over the next 2 weeks. She did not use a hip orthosis. She was also advised to avoid combined hip extension/external rotation maneuvers for the first 4 weeks. She took part in a formal hip-specific physical therapy program for a total of 12 weeks. She was seen in clinic at 2, 6, and 12 weeks postoperatively and appeared to have had a typical, uneventful course. We advised her to gradually return to normal activities as tolerated at the 12-week visit.

Four months after the procedure, the patient returned to our clinic for evaluation after a right hip dislocation. Two days prior, she was at a school function with her child and experienced sudden pain and inability to bear weight after she extended and externally rotated her right hip in a low-energy manner. She was taken to an emergency room and found to have an anterior dislocation of the right hip (Figure 3), which was concentrically reduced under anesthesia.

Upon questioning, she reported having had feelings of mild instability of the right hip during demanding activities (jogging, yoga) after sustaining a low-energy fall 1 month prior to her dislocation. On examination, she had significant apprehension about the right hip during gentle external rotation maneuvers. An MRA 2 weeks after the dislocation showed a large defect of the anterosuperior capsuloligamentous complex measuring 4 cm from medial to lateral and 2.5 cm superior to inferior (Figure 4). No loose bodies, chondral injuries, or recurrent tears of the labrum were seen. Typical postoperative changes were observed at the femoral head-neck junction.

Initially, we recommended nonoperative management with 6 weeks of toe-touch weight-bearing and strict avoidance of hip extension–external rotation maneuvers. No hip orthosis was used. After this period, the patient advanced to full weight-bearing and continued in hip-specific physical therapy. Despite continued therapy and avoidance of provocative maneuvers, the patient reported persistent feelings of right hip instability with significant apprehension during extension and external rotation of the right hip. A repeat MRA 4 months after the hip dislocation showed a persistent defect in the anterosuperior capsuloligamentous complex and no signs of avascular necrosis. After 6 months of conservative treatment, we recommended an open capsulorrhaphy of the right hip with autograft iliotibial band reconstruction of the iliofemoral ligament and capsule.

Six months after the dislocation, the patient underwent the recommended procedure. After induction of general anesthesia, she was placed in the supine position on a standard operating table. A Smith-Petersen approach was used to visualize the anterior hip structures. During deep dissection, we observed a large defect, measuring 2.5×4 cm (Figure 5A), in the anterior hip capsule, with only a thin pseudocapsule covering the femoral head. Extensive mobilization of the anterior capsule was unsuccessful.

The decision was made to harvest a graft from the patient’s ipsilateral iliotibial band. A skin incision was made over the iliotibial band in the distal midthigh region, and a 2.5×4-cm graft was harvested from the central portion of the iliotibial band. An arthrotomy was performed on the hip joint (Figure 5B). The labrum appeared healthy without recurrent tearing or fraying, and other than focal thinning on the superior acetabulum, the cartilage appeared healthy. A double-loaded anchor was placed in the supra-acetabular region, and the sutures were passed through the graft. Then, No. 2 nonabsorbable sutures were sequentially placed between the capsular remnant and the graft medially, inferiorly, and laterally. The graft was placed into position (Figure 5C) and the sutures were tied (Figure 5D).

Postoperatively, the patient was allowed toe-touch weight-bearing for 6 weeks, with strict avoidance of extension–external rotation maneuvers. She participated in a 12-week course of physical therapy with gradual advancement of activities. About a year after the capsulorrhaphy, she was able to resume all previous activities with only occasional low-level discomfort. She returned to the clinic 16 months after the capsulorrhaphy complaining of increased pain with long-distance running but denied feelings of instability. We performed an intra-articular hip injection under ultrasound guidance, which provided 100% relief of her symptoms. We obtained an MRA to evaluate for any recurrent capsular or labral injury (Figure 6). The previous anterosuperior capsular defect was not visible, and no signs of recurrent labral or cartilage injury were seen.

Discussion

With the increasing popularity of hip arthroscopy, more complications are being reported as well, including postoperative hip instability. Three separate cases of anterior hip instability have been published in the past several years.^5-7

Ranawat and colleagues⁵ were the first to report a case of postoperative anterior hip dislocation after arthroscopy. Their patient was a 52-year-old woman with right hip pain and generalized ligamentous laxity. Her preoperative radiographs showed no evidence of degenerative changes, dysplasia, or femoroacetabular impingement. An MRA showed a peripheral tear of the anterosuperior labrum. At arthroscopy, her right hip was easily distracted 2 to 3 cm with what they described as “minimal traction.” A small 1- to 2-cm capsulotomy was performed about the anterior portal. A detached labral tear was identified and repaired with an anchor, and no rim resection was performed. To improve visualization of the peripheral compartment, they extended the previous capsulotomy 1 to 2 cm and débrided the edges. A cam-type lesion was identified and resected. Lastly, they performed an anterior capsular plication, specifically including the iliofemoral ligament. Postoperatively, the patient wore a hip orthosis for 6 weeks to prevent extension and external rotation of the hip as well as a foot brace at night for 3 weeks. The patient was allowed to partially bear weight for the first 6 weeks with use of crutches. Approximately 2 months postoperatively, she slipped and fell down a short flight of stairs. She was diagnosed with an anterior hip dislocation. After successful closed reduction, she was treated conservatively with the same regimen used earlier. She remained symptomatic over the next several months with signs of instability and apprehension, and she eventually underwent a repeat hip arthroscopy. A 1- to 2-cm tear of the anterior capsule and iliofemoral ligament was treated with a revision arthroscopic capsular plication. A postoperative regimen similar to that used at the index procedure was instituted and, at most recent follow-up, she was found to have occasional pain without instability.

Matsuda⁶ reported a case of acute iatrogenic hip dislocation after arthroscopic surgery. His patient was a 39-year-old woman with a mildly retroverted acetabulum leading to impingement about the hip. She had no signs of generalized ligamentous laxity. A hip arthroscopy in the lateral position was performed, with no comment about the extent of the capsulotomy. During the procedure, about 5 mm of anterosuperior acetabulum were removed as part of arthroscopic rim trimming for treatment of the pincer lesion. A femoral osteochondroplasty was also performed (unspecified size) to restore more normal anterolateral offset. One confounding factor was that supranormal hip distraction was needed for 20 minutes to aid in removal of a metallic piece from a radiofrequency ablator, which inadvertently detached. The patient experienced an anterior hip dislocation in the recovery room and was found to be unstable during closed reduction under general anesthesia. A mini-open capsular repair was performed, which showed a 1×1.5-cm defect in the anterolateral capsule. After closure of the defect, the hip was found to be stable under fluoroscopic examination. Postoperatively, the patient was allowed to perform partial weight-bearing in a hip-knee-ankle-foot orthosis for 2 months and then a flexible hip brace for 1 month. At 15-month follow-up, her hip was stable and she was pain-free.

Benali and Katthagen⁷ highlighted the significant contribution of the labrum to hip stability in a dysplastic hip. Their patient was a 49-year-old woman with mild hip dysplasia and a degenerative bucket-handle tear of the ventrolateral labrum. The patient underwent a near-complete labral resection and rim trimming at an outside institution. The patient began full weight-bearing at 3 weeks postoperatively and noticed considerable groin and back pain (no hip orthosis use was mentioned). After failed treatment for suspected lumbar pathology, she was referred to the authors’ clinic for further evaluation. Plain radiographs showed subluxation of the left hip with degenerative changes. The patient had an uneventful left total hip arthroplasty (THA).

After reviewing the 3 reported cases of hip instability after arthroscopy, we suggest that surgeons fully recognize and appreciate the delicate balance of stability and motion provided by the static and dynamic stabilizers of the hip joint, and be cognizant of potential imbalance created by surgical intervention.^8,9 Postarthroscopic hip instability appears to be multifactorial in nature, because all of the reported cases detailed different factors, both patient- and surgeon-related, contributing to instability.

Ranawat and colleagues⁵ identified several factors that may have contributed to the anterior hip dislocation sustained by their patient, including the patient’s generalized ligamentous laxity, performance of a capsulectomy (with repair of iliofemoral ligament), and a traumatic fall. Benali and Katthagen⁷ (although they did not perform the index procedure) described the disastrous complication of overzealous labral resection and rim trimming in a patient with hip dysplasia. Matsuda⁶ performed a labral resection and rim trimming, an extended (unspecified size) capsulotomy, and also used supranormal traction for 20 minutes to remove an iatrogenic foreign body. Surgeons performing hip arthroscopies should be aware of all these factors, because many are directly controlled by the surgeon.

The only factor we feel may have contributed to hip instability in our patient was the performance of a capsulotomy without closure. Our patient was an otherwise healthy woman with no signs of ligamentous laxity, hip dysplasia, or retroversion of the acetabulum. We did not perform a labral resection or rim trimming. We use modified anterior and anterolateral portals, and electrocautery to connect the portals. This typically leads to a release of a thin strip (less than 5 mm wide) of 3 cm of capsule. Based upon findings at rare second-look arthroscopy for recurrent symptoms, Dr. Guanche has observed that the capsulotomy from the initial procedure heals with normal-appearing tissue. Also, during peripheral compartment arthroscopy, we do not routinely release the iliofemoral ligament, and the orbicular ligament is left intact. Instead, we prefer to flex the hip and débride only enough capsular tissue to allow for adequate visualization. 

Little has been published on capsulotomy closure after hip arthroscopy, and no consensus exists. Our standard practice is to not close the capsulotomy, which accords with the practice of other surgeons.⁹ There is concern, however, that extensive capsulotomy leading to injury or disruption of the iliofemoral ligament may cause anterior hip instability, driving other prominent hip arthroscopists to routinely close the capsulotomy.^9,10 Myers and colleagues¹⁰ published a recent biomechanical study on the role of the labrum and the capsular ligaments in hip stability. They concluded that the iliofemoral ligament plays a significant role in limiting external rotation and anterior translation of the femoral head, and recommended closure of the capsulotomy after arthroscopy. Of note, Dr. Guanche has performed more than 1500 hip arthroscopic procedures in the past 5 years, and we are aware of only 2 patients who have sustained anterior hip dislocations, in spite of our not closing the capsulotomy defect. This highlights an important clinical question in need of further investigation.

Our case also raises questions about the ideal postoperative regimen after standard hip arthroscopy. Although we do not routinely prescribe hip orthoses for our patients, others do.⁵ We are unaware of any proven benefit to the standard use of hip orthoses, and are concerned over the possible lack of patient compliance and of adequate restraint. We prefer to educate our patients on avoiding the “at-risk” position of hip extension and external rotation and to counsel them on gradual return to activities. Studies are needed to determine the role of these devices in hip arthroscopy, as well as the ideal postoperative activity regimen.

Our patient failed 6 months of conservative treatment after her dislocation and continued to have feelings of hip instability even during light activities. As a result of this failure and given an anatomical defect in the anterior capsuloligamentous complex, we decided our patient would be best treated with reconstruction of the defect. We did not think a revision capsular plication, as done by Ranawat and colleagues,⁵ was a reasonable option for our patient because of a large defect in the capsular tissue. Even in smaller defects, plication could potentially lead to overtightening of the capsule and subsequent overconstraint of the joint. Also, plication of defects may place excessive strain on the suture, which may fail if the repair is even mildly stressed.

Recurrent anterior hip dislocations, although rare in their own right, are much more common after THA than after hip arthroscopy.¹¹ Fujishiro and colleagues¹² described a similar technique to ours developed to treat a patient with recurrent anterior hip instability from anterior capsular insufficiency after multiple revision THA procedures. They used a Leeds-Keio artificial ligament to reconstruct the iliofemoral ligament, and this successfully treated their patient’s instability.

Conclusion

We believe this is the first report of recurrent instability after hip arthroscopy, necessitating reconstruction of the anterior capsuloligamentous complex. This case shows that reconstruction of the iliofemoral ligament with iliotibial band autograft is safe, restores hip stability without compromising function, and should be considered by any hip arthroscopist encountering a similar scenario. It also highlights the importance of the capsuloligamentous complex surrounding the hip joint for its stability and the need for further research to better delineate the indications for capsular repair/closure after capsulotomy.

THE CASE

A 37-year-old right-hand dominant woman came to our clinic seeking treatment for bilateral generalized hand cramping and weakness that she had been experiencing for approximately 2 to 3 years. She was dropping objects and had finger locking, yet had no numbness, tingling, or morning stiffness.

Ten months earlier, she had given birth to a healthy 3715 g girl. Our patient’s prenatal glucose tolerance test had been normal. Her pregnancy and delivery had been significant for oligohydramnios, failed post-term (41 weeks 4 days) induction, and emergent low transverse cesarean section due to fetal bradycardia. Since giving birth, our patient had 3 menstrual periods while breastfeeding. She had a copper intrauterine device inserted at her 6-week postpartum visit. She also had 2 truncal acrochordons removed 3 months postpartum. She had no history of neck trauma, overuse injury, or occupational exposures.

Her blood pressure and vital signs were within normal limits. Physical exam was notable for subtly coarse facial features and broad fingers (FIGURE 1).

She had normal wrist and hand joint range of motion; her wrist and hand strengths, including grip strength, were 5 out of 5. Tinel’s sign, Phalen’s maneuver, and Finkelstein’s test were negative.

Her upper extremity neurovascular exams were completely normal. Initial laboratory studies—including a comprehensive metabolic panel—were normal. The only exception was her creatine kinase, which was
265 U/L (normal, 24-195 U/L).

At a follow-up appointment 7 weeks later, we gathered a more detailed history and learned that over the past 2 to 3 years, the patient had noticed that her shoe and ring sizes had been increasing. She also mentioned some mild weight gain following her pregnancy.

Occasionally, she had generalized hand swelling, headaches, and saw floaters, but she denied losing peripheral vision. Additional lab work at this time revealed a fasting growth hormone (GH) level of 27.3 ng/mL (normal, 0.05-8 ng/mL) and an insulin-like growth factor 1 (IGF-1) level of 848 ng/mL (normal, 106-368 ng/mL). An anterior pituitary hormone panel and cortisol level were normal. A urine pregnancy test was negative.

THE DIAGNOSIS

Magnetic resonance imaging (MRI) of our patient’s brain revealed a pituitary adenoma (FIGURE 2). Based on that and the patient’s elevated GH and IGF-1 levels, we diagnosed acromegaly due to a pituitary adenoma.

DISCUSSION

Acromegaly is a rare, progressively disfiguring disease with a prevalence of 40 cases per million people.¹ It affects middle-aged adults, with no gender difference.² In most cases, the cause is a benign pituitary adenoma.^1-4

Patients with acromegaly may complain
 of carpal tunnel-like symptoms, headaches, and visual disturbances. Physical changes include coarse facial features, generalized expansion of the skull, brow protrusion, ocular distension, prognathism, macroglossia, acral overgrowth, and dental malocclusion; these changes typically occur slowly over a long time period.^1-5 For example, when we looked at the 3-year-old photo on our patient’s driver’s license, we noticed only subtle changes from her current appearance. Common clinical manifestations include headache, hyperpigmentation, hypertrichosis, hyperhidrosis, goiter, arthropathy, carpal tunnel syndrome, visual disturbances, and acrochordons.^1,5

Acromegaly is associated with an increased risk of cardiovascular disease, metabolic disorders, infertility, sleep apnea, arthritis, thyroid tumors, colon adenomas, and carcinoma.^1,2,4,5 Due to the insidious progression of acromegaly’s clinical manifestations, diagnosis is delayed for 4 to 10 years, on average.¹ The diagnosis of acromegaly is typically based on an elevation of GH and IGF-1 levels.^1,5 A brain MRI is essential in the diagnosis of a pituitary adenoma.¹

Pregnancy among patients with acromegaly is uncommon. In fact, fewer than 150 cases have been reported in the literature.^2,6 In most cases, it appears that pregnancy among patients with acromegaly is safe for mothers and newborns.^6,7

The goals of treatment for acromegaly caused by a pituitary adenoma are to remove/ reduce the tumor and its mechanical effects, relieve symptoms, reduce serum GH and IGF-1, and restore pituitary function. Transsphenoidal surgical resection is the preferred treatment for pituitary adenomas.^1,2,4 Radiation therapy and pharmacologic treatment may be necessary as adjuncts to surgery or for patients for whom surgery is contraindicated.^1,4,5

Pharmacologic management of acromegaly includes dopamine agonists (cabergoline), somatostatin analogues (octreotide, lanreotide), and GH receptor antagonists (pegvisomant).^1,3 Patients who receive effective early treatment of acromegaly have a life expectancy similar to that of the general population.^1,5

Our patient

Our patient was referred to Neurosurgery and underwent transnasal transsphenoidal resection of the pituitary adenoma. Two weeks postop, her GH level had decreased to 0.66 ng/mL and her IGF-1 level was down to 386 ng/mL. Four months later, her GH (2.32 ng/mL) and IGF-1 levels (277 ng/mL) were within normal range and our patient reported improvement in all of her symptoms.

THE TAKEAWAY

Because it may take years for the classical clinical features of acromegaly such as coarse facial features, protruding jaw, and broad fingers to become apparent, diligent history taking is essential to diagnose the condition early. Patients may present with nonspecific and confusing symptoms such as muscle weakness.⁸ Early nonspecific symptoms and signs in the presence of normal basic laboratory tests should warrant an evaluation of fasting GH and IGF-1. Early treatment with surgery, radiation therapy, or pharmacotherapy may prevent or decrease the intensity of rheumatologic, cardiovascular, respiratory, and metabolic complications of acromegaly.¹

THE CASE

A 37-year-old right-hand dominant woman came to our clinic seeking treatment for bilateral generalized hand cramping and weakness that she had been experiencing for approximately 2 to 3 years. She was dropping objects and had finger locking, yet had no numbness, tingling, or morning stiffness.

Ten months earlier, she had given birth to a healthy 3715 g girl. Our patient’s prenatal glucose tolerance test had been normal. Her pregnancy and delivery had been significant for oligohydramnios, failed post-term (41 weeks 4 days) induction, and emergent low transverse cesarean section due to fetal bradycardia. Since giving birth, our patient had 3 menstrual periods while breastfeeding. She had a copper intrauterine device inserted at her 6-week postpartum visit. She also had 2 truncal acrochordons removed 3 months postpartum. She had no history of neck trauma, overuse injury, or occupational exposures.

Her blood pressure and vital signs were within normal limits. Physical exam was notable for subtly coarse facial features and broad fingers (FIGURE 1).

She had normal wrist and hand joint range of motion; her wrist and hand strengths, including grip strength, were 5 out of 5. Tinel’s sign, Phalen’s maneuver, and Finkelstein’s test were negative.

Her upper extremity neurovascular exams were completely normal. Initial laboratory studies—including a comprehensive metabolic panel—were normal. The only exception was her creatine kinase, which was
265 U/L (normal, 24-195 U/L).

At a follow-up appointment 7 weeks later, we gathered a more detailed history and learned that over the past 2 to 3 years, the patient had noticed that her shoe and ring sizes had been increasing. She also mentioned some mild weight gain following her pregnancy.

Occasionally, she had generalized hand swelling, headaches, and saw floaters, but she denied losing peripheral vision. Additional lab work at this time revealed a fasting growth hormone (GH) level of 27.3 ng/mL (normal, 0.05-8 ng/mL) and an insulin-like growth factor 1 (IGF-1) level of 848 ng/mL (normal, 106-368 ng/mL). An anterior pituitary hormone panel and cortisol level were normal. A urine pregnancy test was negative.

THE DIAGNOSIS

Magnetic resonance imaging (MRI) of our patient’s brain revealed a pituitary adenoma (FIGURE 2). Based on that and the patient’s elevated GH and IGF-1 levels, we diagnosed acromegaly due to a pituitary adenoma.

DISCUSSION

Acromegaly is a rare, progressively disfiguring disease with a prevalence of 40 cases per million people.¹ It affects middle-aged adults, with no gender difference.² In most cases, the cause is a benign pituitary adenoma.^1-4

Patients with acromegaly may complain
 of carpal tunnel-like symptoms, headaches, and visual disturbances. Physical changes include coarse facial features, generalized expansion of the skull, brow protrusion, ocular distension, prognathism, macroglossia, acral overgrowth, and dental malocclusion; these changes typically occur slowly over a long time period.^1-5 For example, when we looked at the 3-year-old photo on our patient’s driver’s license, we noticed only subtle changes from her current appearance. Common clinical manifestations include headache, hyperpigmentation, hypertrichosis, hyperhidrosis, goiter, arthropathy, carpal tunnel syndrome, visual disturbances, and acrochordons.^1,5

Acromegaly is associated with an increased risk of cardiovascular disease, metabolic disorders, infertility, sleep apnea, arthritis, thyroid tumors, colon adenomas, and carcinoma.^1,2,4,5 Due to the insidious progression of acromegaly’s clinical manifestations, diagnosis is delayed for 4 to 10 years, on average.¹ The diagnosis of acromegaly is typically based on an elevation of GH and IGF-1 levels.^1,5 A brain MRI is essential in the diagnosis of a pituitary adenoma.¹

Pregnancy among patients with acromegaly is uncommon. In fact, fewer than 150 cases have been reported in the literature.^2,6 In most cases, it appears that pregnancy among patients with acromegaly is safe for mothers and newborns.^6,7

The goals of treatment for acromegaly caused by a pituitary adenoma are to remove/ reduce the tumor and its mechanical effects, relieve symptoms, reduce serum GH and IGF-1, and restore pituitary function. Transsphenoidal surgical resection is the preferred treatment for pituitary adenomas.^1,2,4 Radiation therapy and pharmacologic treatment may be necessary as adjuncts to surgery or for patients for whom surgery is contraindicated.^1,4,5

Pharmacologic management of acromegaly includes dopamine agonists (cabergoline), somatostatin analogues (octreotide, lanreotide), and GH receptor antagonists (pegvisomant).^1,3 Patients who receive effective early treatment of acromegaly have a life expectancy similar to that of the general population.^1,5

Our patient

Our patient was referred to Neurosurgery and underwent transnasal transsphenoidal resection of the pituitary adenoma. Two weeks postop, her GH level had decreased to 0.66 ng/mL and her IGF-1 level was down to 386 ng/mL. Four months later, her GH (2.32 ng/mL) and IGF-1 levels (277 ng/mL) were within normal range and our patient reported improvement in all of her symptoms.

THE TAKEAWAY

Because it may take years for the classical clinical features of acromegaly such as coarse facial features, protruding jaw, and broad fingers to become apparent, diligent history taking is essential to diagnose the condition early. Patients may present with nonspecific and confusing symptoms such as muscle weakness.⁸ Early nonspecific symptoms and signs in the presence of normal basic laboratory tests should warrant an evaluation of fasting GH and IGF-1. Early treatment with surgery, radiation therapy, or pharmacotherapy may prevent or decrease the intensity of rheumatologic, cardiovascular, respiratory, and metabolic complications of acromegaly.¹

THE CASE

A 37-year-old right-hand dominant woman came to our clinic seeking treatment for bilateral generalized hand cramping and weakness that she had been experiencing for approximately 2 to 3 years. She was dropping objects and had finger locking, yet had no numbness, tingling, or morning stiffness.

Ten months earlier, she had given birth to a healthy 3715 g girl. Our patient’s prenatal glucose tolerance test had been normal. Her pregnancy and delivery had been significant for oligohydramnios, failed post-term (41 weeks 4 days) induction, and emergent low transverse cesarean section due to fetal bradycardia. Since giving birth, our patient had 3 menstrual periods while breastfeeding. She had a copper intrauterine device inserted at her 6-week postpartum visit. She also had 2 truncal acrochordons removed 3 months postpartum. She had no history of neck trauma, overuse injury, or occupational exposures.

Her blood pressure and vital signs were within normal limits. Physical exam was notable for subtly coarse facial features and broad fingers (FIGURE 1).

She had normal wrist and hand joint range of motion; her wrist and hand strengths, including grip strength, were 5 out of 5. Tinel’s sign, Phalen’s maneuver, and Finkelstein’s test were negative.

Her upper extremity neurovascular exams were completely normal. Initial laboratory studies—including a comprehensive metabolic panel—were normal. The only exception was her creatine kinase, which was
265 U/L (normal, 24-195 U/L).

At a follow-up appointment 7 weeks later, we gathered a more detailed history and learned that over the past 2 to 3 years, the patient had noticed that her shoe and ring sizes had been increasing. She also mentioned some mild weight gain following her pregnancy.

Occasionally, she had generalized hand swelling, headaches, and saw floaters, but she denied losing peripheral vision. Additional lab work at this time revealed a fasting growth hormone (GH) level of 27.3 ng/mL (normal, 0.05-8 ng/mL) and an insulin-like growth factor 1 (IGF-1) level of 848 ng/mL (normal, 106-368 ng/mL). An anterior pituitary hormone panel and cortisol level were normal. A urine pregnancy test was negative.

THE DIAGNOSIS

Magnetic resonance imaging (MRI) of our patient’s brain revealed a pituitary adenoma (FIGURE 2). Based on that and the patient’s elevated GH and IGF-1 levels, we diagnosed acromegaly due to a pituitary adenoma.

DISCUSSION

Acromegaly is a rare, progressively disfiguring disease with a prevalence of 40 cases per million people.¹ It affects middle-aged adults, with no gender difference.² In most cases, the cause is a benign pituitary adenoma.^1-4

Patients with acromegaly may complain
 of carpal tunnel-like symptoms, headaches, and visual disturbances. Physical changes include coarse facial features, generalized expansion of the skull, brow protrusion, ocular distension, prognathism, macroglossia, acral overgrowth, and dental malocclusion; these changes typically occur slowly over a long time period.^1-5 For example, when we looked at the 3-year-old photo on our patient’s driver’s license, we noticed only subtle changes from her current appearance. Common clinical manifestations include headache, hyperpigmentation, hypertrichosis, hyperhidrosis, goiter, arthropathy, carpal tunnel syndrome, visual disturbances, and acrochordons.^1,5

Acromegaly is associated with an increased risk of cardiovascular disease, metabolic disorders, infertility, sleep apnea, arthritis, thyroid tumors, colon adenomas, and carcinoma.^1,2,4,5 Due to the insidious progression of acromegaly’s clinical manifestations, diagnosis is delayed for 4 to 10 years, on average.¹ The diagnosis of acromegaly is typically based on an elevation of GH and IGF-1 levels.^1,5 A brain MRI is essential in the diagnosis of a pituitary adenoma.¹

Pregnancy among patients with acromegaly is uncommon. In fact, fewer than 150 cases have been reported in the literature.^2,6 In most cases, it appears that pregnancy among patients with acromegaly is safe for mothers and newborns.^6,7

The goals of treatment for acromegaly caused by a pituitary adenoma are to remove/ reduce the tumor and its mechanical effects, relieve symptoms, reduce serum GH and IGF-1, and restore pituitary function. Transsphenoidal surgical resection is the preferred treatment for pituitary adenomas.^1,2,4 Radiation therapy and pharmacologic treatment may be necessary as adjuncts to surgery or for patients for whom surgery is contraindicated.^1,4,5

Pharmacologic management of acromegaly includes dopamine agonists (cabergoline), somatostatin analogues (octreotide, lanreotide), and GH receptor antagonists (pegvisomant).^1,3 Patients who receive effective early treatment of acromegaly have a life expectancy similar to that of the general population.^1,5

Our patient

Our patient was referred to Neurosurgery and underwent transnasal transsphenoidal resection of the pituitary adenoma. Two weeks postop, her GH level had decreased to 0.66 ng/mL and her IGF-1 level was down to 386 ng/mL. Four months later, her GH (2.32 ng/mL) and IGF-1 levels (277 ng/mL) were within normal range and our patient reported improvement in all of her symptoms.

THE TAKEAWAY

Because it may take years for the classical clinical features of acromegaly such as coarse facial features, protruding jaw, and broad fingers to become apparent, diligent history taking is essential to diagnose the condition early. Patients may present with nonspecific and confusing symptoms such as muscle weakness.⁸ Early nonspecific symptoms and signs in the presence of normal basic laboratory tests should warrant an evaluation of fasting GH and IGF-1. Early treatment with surgery, radiation therapy, or pharmacotherapy may prevent or decrease the intensity of rheumatologic, cardiovascular, respiratory, and metabolic complications of acromegaly.¹