Bladder perforation by orthopedic implants 26 years after limb-sparing surgery for left proximal femoral chondrosarcoma: A case report.

INTRODUCTION: The need for implant use during orthopedic surgeries has been increasing. Accordingly, increased implant failures have been reported. However, bladder perforation remains a rare complication after orthopedic surgery. Although a few reports have described bladder perforation after total hip arthroplasty, no previous studies have reported the migration of staples into the bladder after limb-sparing surgery. PRESENTATION OF CASE: A 65-year-old patient underwent limb-sparing surgery to remove a chondrosarcoma in the left proximal thigh. Twenty-six years after surgery, a staple that had been used to fix artificial ligaments to the pubis migrated to perforate the bladder, resulting in painful urination. The staple was removed, and her symptoms improved. DISCUSSION: In this case, bladder perforation by the staple resulted in painful urination. The bladder perforation was not detected until 26 years after the initial surgery. CONCLUSION: Our observations emphasize that implant complications may occur even after a long postoperative period, and the possibility of delayed bladder perforation from previous pelvic surgeries should be considered in patients presenting with urinary tract symptoms.

Osteofibrous dysplasia arising in the humerus: A case report.

Osteofibrous dysplasia is a benign fibro-osseous lesion of bone which is most commonly occurred in cortical bone of anterior mid-shaft of the tibia of infancy and childhood. This study reported a case of osteofibrous dysplasia arising in the humerus of adult, resulting in good prognosis after a surgical treatment. A 34-year-old male had felt left upper arm pain and was suspected as having a bone tumor at the humeral shaft by X-ray pictures. The tumor was suspected as the osteofibrous dysplasia of the humerus by a core needle biopsy. Intralesional curettage, intraoperative anhydrous ethanol therapy, and artificial bone graft were performed. Surgical specimens showed fibro-osseous lesion, which strongly indicated osteofibrous dysplasia. Seven years after the surgery, he has lived without any local recurrence and complaints. It is important to recognize that osteofibrous dysplasia can arise in the humerus of an older patient for appropriate diagnosis.

Clinical features and radiological findings of 67 patients with SAPHO syndrome.

OBJECTIVES: The purpose of this study was to facilitate the understanding of the SAPHO (Synovitis, Acne, Pustulosis, Hyperostosis, and Osteitis) syndrome by analyzing the clinical and radiological features of 67 Japanese patients with SAPHO syndrome. METHODS: Sixty-seven Japanese patients (female/male: 44/23, mean age at onset: 48.5 years) were diagnosed with SAPHO syndrome from 2002 to 2013 at our hospital. Medical records and radiological imaging of these patients were retrospectively reviewed. RESULTS: Among the 67 patients, 41 had dermatological manifestations, such as palmoplantar pustulosis, acne, and psoriasis. Initial symptom was local pain in all patients, and the most common initial site of the symptom was the anterior chest. Bacterial and fungal cultures from 20 bone biopsies were all negative. Histopathological diagnosis of the specimens was non-specific inflammation in all cases. Bone lesions were observed in 65 patients (97.0%). On the other hand, articular lesions including enthesitis were found in 31 patients (46.2%). CONCLUSION: SAPHO syndrome had different clinical and radiological aspects. The clinical features were not remarkable, except the dermatological manifestations and the involvement of the anterior chest. Bone lesions including hyperostosis and osteitis were found radiographically in the majority of patients with SAPHO syndrome. These are the characteristics of the SAPHO syndrome, with the exclusion of other bone diseases.

Prediction of pathological fracture of the femoral shaft with an osteolytic lesion using a computed tomography-based nonlinear three-dimensional finite element method.

BACKGROUND: Physicians radiologically estimate the reduction in bone strength based on the size or location of bone tumors. The goal of this study was to clarify the relationship between the size or location of a bony defect and its mechanical strength using a computed tomography-based three-dimensional finite element method. METHODS: Computed tomography data of the right femur from two volunteers (one healthy male and one female patient with primary osteoporosis) were used for the present study. A spherical defect of various sizes and locations at the level of the isthmus of the femoral shaft was created on the three-dimensional finite element models to simulate the osteolytic bone tumor. We classified these defects into three types: inner erosion, cortical disruption, and outer erosion. Two types of mechanical testing were performed: axial compression and torsion. RESULTS: In the axial compression testing of the healthy male subject, the correlation coefficients between the defect rate and the failure load in the cortical disruption type, inner erosion type, and outer erosion type were -0.916, -0.358, and -0.106, respectively. In the torsion testing, they were -0.8744, -0.9001, and -0.8907, respectively. In the axial compression testing of the osteoporotic female subject, the correlation coefficients in the cortical disruption type, inner erosion type, and outer erosion type were -0.754, -0.621, and -0.158, respectively. In the torsion testing, they were -0.9199, -0.5098, and -0.8363, respectively. In both tests, the defect rate of the cortex increased and the bone strength decreased, especially in the cortical disruption type. CONCLUSION: The results of the present study demonstrate that osteolytic bone tumors can weaken the bone strength, particularly when perforation of the cortex occurs via tumor invasion. These results may be useful for risk assessment of pathological fractures due to primary and metastatic osteolytic bone tumors in clinical practice.

Sex steroid synthesis in human skin in situ: the roles of aromatase and steroidogenic acute regulatory protein in the homeostasis of human skin.

Sex steroids have been known to play important roles in the homeostasis of human skin, but little is known about their biosyntheses in that tissue. In this study, we characterized the correlation between the concentrations of sex steroids and the expression levels of the factors involved in their synthesis or metabolism in human skin. The expression levels of aromatase (ARO) and steroidogenic acute regulatory protein (StAR) were positively correlated with estrogens and testosterone concentrations, respectively. We demonstrated that estrogen synthesis was markedly decreased by ARO inhibitor and that skins with higher ARO expression had thicker elastic fibers than those with lower ARO expression. While pregnenolone and testosterone concentrations were increased by cholesterol administration to epidermal keratinocytes. Scalp skin with higher StAR expression was cleared to have significantly fewer hair follicles than that with lower expression. Our results suggest that the status of ARO and StAR contribute to estrogen synthesis in situ, especially for the regulation of elastic fiber formation, and to testosterone synthesis, which may be associated with hair growth, respectively.

1α,25-dihydroxyvitamin D3 enhances fast-myosin heavy chain expression in differentiated C2C12 myoblasts.

We investigated the effect of VD3 (1α,25-dihydroxyvitamin D3) on the proliferating, differentiating and differentiated phases of C2C12 myoblasts, a mouse skeletal muscle cell line. VD3 treatment in 10% FBS (fetal bovine serum) inhibited the proliferation and viability of the cells in a dose-dependent manner. It also dose-dependently increased the percentage of cells in the G0/G1 phase as shown by flow cytometry. In the differentiating phase, VD3 treatment inhibited the formation of myotubes and the expression of total myosin heavy chain at both the mRNA and protein levels. In the differentiated phase, treatment had no significant effect on the amount of total myosin heavy chain, as Western blot analysis with MF20 antibody [DSHB (Developmental Studies Hybridoma Bank)] showed. However, significantly greater expression of fast myosin heavy chain in 1 nM VD3 was found by Western blot analysis with MY-32 (Sigma). Thus VD3 inhibited the proliferation of myoblasts during proliferating and differentiating phases, whereas it increased the expression of the fast myosin heavy chain isoform in the differentiated phase. The data indicate that an adequate concentration of VD3 might have an anabolic effect on differentiated skeletal muscle.

Painful extraskeletal osteochondroma under the tarsal sesamoid: a case report and review of literature.

Extraskeletal osteochondroma is a rare condition. To our knowledge, occurrence in the foot has been very rare. We present here a case wherein the patient developed a painful extraskeletal osteochondroma under the tarsal sesamoid. A fifty-one-year-old man presented at our department with a painful mass on the plantar part of the metatarsal joint of the right big toe. The mass had grown gradually in size for the past 7 months and was associated with pain while walking. Preoperative X-ray and a computed tomography showed that the tumor had no continuity with adjacent bone. Intraoperatively, a bony hard tumor was located in the subcutaneous tissue plantar to the tibial sesamoid. The mass was firmly attached to the sheath of the flexor hallucis brevis tendon. Macroscopically, the tumor was covered with glistering surface. The tumor was carefully dissected from the surrounding soft tissues and was removed. It was diagnosed as extraskeletal osteochondroma histologically. The patient had no recurrence at 1 year after operation.

The role of estrogen-metabolizing enzymes and estrogen receptors in human epidermis.

Local estrogen metabolism and its sensitivities in the skin have been also suggested to contribute to skin homeostasis in addition to age- and/or gender-dependent circulating estrogen, even though their local mechanisms have been largely unknown. To characterize their potential correlations, age- and gender-dependencies were evaluated focusing on 5 pivotal estrogen-metabolizing enzymes including aromatase, estrogen sulfotransferase, steroid sulfatase, and 17ß-hydroxysteroid dehydrogenases and estrogen receptors (ERα and ERß) using immunohistochemistry of 100 human skin specimens. When their epidermal expression levels were compared among 7 age groups, ranging from the teens to the seventies, the highest expression in the teens group and the lowest expression in the seventies group were found in the expression of aromatase and ERß, respectively, while no significant differences between the male and the female groups were found in the immunoreactivities of our interested proteins. Our results suggest that age-related differences in aromatase and ERß expressions impact epidermal homeostasis.

Tumors masked as frozen shoulders: a retrospective analysis.

BACKGROUND: It was reported that some shoulder tumors were misdiagnosed with frozen shoulder syndrome. The purposes of this study were to elucidate the incidence of the initial misdiagnosis with frozen shoulder syndrome among the patients with malignant shoulder tumors, and to clarify whether such initial misdiagnosis affected the time to make a final correct diagnosis or not. METHODS: Clinical records of 34 patients (age>40) with malignant shoulder tumors and those of 505 patients (age>40) with shoulder pain and stiffness were reviewed in the author's institute. The duration of the prediagnostic period was compared between the patients with and without an initial misdiagnosis as frozen shoulder syndrome. RESULTS: Among 34 tumor patients, 9 (26%) had been initially misdiagnosed with frozen shoulder syndrome. Two patients actually manifested shoulder pain and stiffness, although they did not have a record of misdiagnosis. Among 505 patients with shoulder pain and stiffness, 4 (0.8%) were diagnosed later as having malignant tumors. One of these 4 patients had been initially misdiagnosed with frozen shoulder syndrome. Consequently, 15 malignant tumors (10 bone tumors and 5 soft tissue sarcomas) were identified. Seven of them were intraosseous humeral tumors and 4 were localized in the scapular region, where patients themselves could not find them. In 10 patients, initial misdiagnosis as frozen shoulder syndrome did cause a significant delay to reach the correct diagnosis as malignant tumors (P=.035). CONCLUSION: Physicians should carefully re-examine the frozen shoulder patients with repeated plain radiographs followed by further imaging studies, if the conservative therapy fails.

Secondary osteosarcoma arising after treatment for childhood hematologic malignancies.

Secondary osteosarcoma arising after the treatment of hematologic malignancies other than Hodgkin's lymphoma is rare. We report two cases of secondary osteosarcoma arising after treatment for childhood hematologic malignancies (non-Hodgkin's lymphoma and lymphoblastic leukemia). A 10-year-old boy, at the age of 3, was diagnosed with non-Hodgkin's lymphoma. He received chemotherapy, radiation, and bone-marrow transplantation and then was in complete remission. At 6 years, he complained of increasing pain of the right thigh and was diagnosed with osteoblastic osteosarcoma. A 26-year-old man, at the age of 6, was diagnosed as having acute lymphoblastic leukemia (ALL). He received chemotherapy, radiation, and peripheral blood stem cell transplantation (PBSCT). At 11 years after PBSCT, he visited with the complaint of left lumbar swelling. He was diagnosed with chondroblastic osteosarcoma. In both cases alkaline phosphatase (ALP) had already increased prior to the onset of the symptom. We should rule out secondary osteosarcoma at the abnormal elevation of ALP during clinical follow-up of patients after treatment of childhood hematologic malignancies.

Histogenesis-specific expression of fibroblast activation protein and dipeptidylpeptidase-IV in human bone and soft tissue tumours.

AIMS: Fibroblast activation protein (FAP)/seprase and dipeptidylpeptidase-IV (DPP-IV)/CD26 are serine integral membrane proteases. They are involved in tissue remodelling, cancer invasion and metastases, mechanisms that are controversial. The aim was to identify cell types that express FAP and DPP-IV in human bone and soft tissue tumours, and to determine whether there are any correlations between the expression of FAP and DPP-IV and the malignant potential of tumours. METHODS AND RESULTS: This study analysed in situ expression in 25 malignant and 13 benign human bone and soft tissue tumours. Reverse transcriptase-polymerase chain reaction analyses confirmed mRNA expression of FAP and DPP-IV in all individuals. Immunohistochemistry using pre-fixed frozen sections revealed that FAP was positive in low-grade myofibroblastic sarcoma, the fibroblastic component of osteosarcomas, and malignant fibrous histiocytomas, but negative in Ewing's sarcomas and rhabdomyosarcomas. DPP-IV showed similar immunohistochemical results. Among benign tumours, non-ossifying fibromas, desmoid tumours and chondroblastomas expressed both FAP and DPP-IV. Giant cells expressed DPP-IV in giant cell tumours. CONCLUSIONS: Our data suggest that FAP and DPP-IV are consistently expressed in bone and soft tissue tumour cells that are histogenetically related to activated fibroblasts and/or myofibroblasts, irrespective of their malignancy. DPP-IV is also expressed in monocyte-macrophage lineage cells.

Calcific myonecrosis and the role of imaging in the diagnosis: a case report.

Calcific myonecrosis is a rare posttraumatic complication characterized by the replacement of muscles of one or more compartments with central liquefaction and peripheral calcification. We report magnetic resonance imaging (MRI) and CT imaging features of calcific myonecrosis arising 43 years after trauma. A 62-year-old man presented with a slowly enlarging mass in the left lower leg. Plain radiographs revealed a soft tissue mass with extensive amorphous calcification. The middle third of the tibia and fibula were eroded. MRI demonstrated peripheral ring enhancement on postcontrast fat-suppressed T1-weighted images. Volume-rendered images extracting only the images of bone and vessels also showed many branches from the tibialis posterior and peroneal arteries around the bone defect. During the operation, bleeding continued heavily from the vessels penetrating the cortical bone of the tibia, from the posterior compartment, and from the branches of tibialis posterior artery. The total blood loss was approximately 2,400 milliliters. There may be a causal relationship between massive bleeding and the hypervascularity of this tumor as evidenced by MRI and volume-rendering CT studies.

Multiple bone metastasis of sclerosing epithelioid fibrosarcoma 12 years after initial surgery-increasing ki-67 labeling index.

Sclerosing epithelioid fibrosarcoma (SEF) is a rare sarcoma of low-grade malignancy. There has been no report to describe the comparison of histological features of SEF between primary and metastatic lesions in spite of high local recurrence rate. We report the histological changes and increasing Ki-67 labeling index of the primary and metastatic lesions of SEF. The patient was a 31-year-old man. At 18, a tumor in the abdominal wall was excised. At 23, the tumor recurred which was removed again. At 30, he was referred to our hospital because of swelling and pain in the chest. Histological examination of the chest wall tumor showed epithelioid cells arranged like alveolar pattern with dense collagen stroma. These findings were consistent with those of SEF. Abdominal and the rib tumors showed the same immunohistochemistrical expression. It is noteworthy that the tumor cells of the rib lesion showed increased cellularity, and its Ki-67 activity was higher as compared with the abdominal tumor, suggestive of progression of malignancy of SEF.

Effects of tendon transfer to restore index finger abduction for severe cubital tunnel syndrome.

BACKGROUND: Severe cases of cubital tunnel syndrome do not always result in functional recovery after surgical decompression of the ulnar nerve. A combined operation of tendon transfer to restore index finger abduction and decompression of the ulnar nerve was performed for patients with severe cubital tunnel syndrome who required powerful pinch strength and whose preoperative compound muscle action potential of the abductor digiti minimi muscle was not recordable or almost non-recordable. The purpose of this study was to assess the efficacy of tendon transfer to restore index finger abduction for severe cubital tunnel syndrome. METHODS: Sixteen hands in 15 patients with severe cubital tunnel syndrome were operated on with extensor pollicis brevis tendon transfer to the first dorsal interosseous muscle to restore index finger abduction and ulnar nerve decompression. They were reviewed after a mean follow-up of 16 months. All 16 hands had preoperatively severe lesions with paralysis of ulnar intrinsics, marked anaesthesia, or hypaesthesia. RESULTS: Postoperative results were excellent in 2 hands, good in 10, fair in 4, and no cases with poor results according to Akahori's criteria. Four hands with fair results had a residual Froment sign or annoying hypaesthesia in the ring and little fingers. All patients were relieved of preoperative discomfort and showed recovery of motor and sensory function. The mean pre- and postoperative pinch strength was 3.3 kg and 5.6 kg, respectively, which showed a significant difference (P<0.01). Mean time of showing a negative Froment sign after surgery was 5 months in 13 cases. CONCLUSIONS: The extensor pollicis brevis tendon transfer is simple and useful to restore index finger abduction and pinch strength for severe cubital tunnel syndrome.

Imaging features of foreign body granuloma in the lower extremities mimicking a soft tissue neoplasm.

Foreign body granuloma is a tissue reaction for retained foreign bodies after skin-penetrating trauma. Detection of retained foreign bodies can be extremely difficult when the patients present with non-specific symptoms such as pain and/or swelling without recognizing a previous trauma. We report three patients of foreign body granulomas in the lower extremities with emphasis placed on their unique clinical and radiological features. The involved sites were the foot, posterior thigh, and posterior lower leg, with wooden splinters in two patients and a fragment of tile in one. Plain radiographs could not reveal the existence of foreign bodies. Magnetic resonance imaging (MRI) showed foreign bodies as low intensities on both T1- and T2-weighted images in two patients, and the surrounding reactive lesion as low to iso intensities on T1- and high intensities on T2-weighted images in all the patients. The peripheral areas of the lesion were strongly enhanced after gadolinium injection. Ultrasound sonography could clearly visualize a foreign body as an echogenic area with posterior acoustic shadowing in one patient. The surrounding ring-like reactive lesion is easily mistaken for a soft tissue neoplasm when foreign bodies are not identified. The key to arriving at the correct diagnosis is to clarify the previous trauma and to identify foreign bodies with low signal intensities on both T1- and T2-weighted images and/or the characteristic ring-like enhancement on MRI. It is also necessary to rule out a foreign body granuloma whenever we see patients with a soft tissue tumor in the extremities, irrespective of their previous trauma history.

A patient with focal fibrocartilaginous dysplasia in the distal femur and review of the literature.

Focal fibrocartilaginous dysplasia (FFCD) is a rare and benign bone lesion that induces bowing deformity of the long bones in young children. Excessive production of fibrocartilage by abnormal differentiation in the metaphysis or by trauma during delivery or after birth is thought to cause growth disturbance. Radiologically, the lesion is characterized by a lucent defect with marginal sclerosis in the medial metaphysis of the long bone. However, there have been few reports about the initial radiological changes of FFCD before bowing started. We report a patient with FFCD in the left distal femur in whom the radiological changes were serially observed during the course of the disorder. A 2-week-old boy first visited our hospital because of left thigh pain. Plain radiographs did not show any abnormal findings at that time. At 10 weeks, a well-defined lucent defect with bony fragment inside was observed in the distal femoral medial cortex. At 1 year, this bony fragment gradually vanished but varus deformity progressed and reached approximately 40 degrees at the age of 2. After removal of the lesion, osteotomy and immobilization was performed with Ilizarov external fixator comprising rings, rods and wires. Complete bone union was achieved 3 months after operation. It is noteworthy that we could observe the initial radiological changes of FFCD before varus deformity occurred. As far as we know, there have been no descriptions of the bony fragment inside a lucent defect of the lesion. Radiological features may vary in the early phase of FFCD.

Open elbow arthrolysis for post-traumatic elbow contracture.

BACKGROUND: Post-traumatic contracture is a common complication after elbow trauma. If conservative therapy fails to restore adequate elbow motion, arthrolysis is indicated. The purposes of this study were to evaluate the clinical outcome of open arthrolysis for post-traumatic elbow contracture and to determine factors influencing the outcome. METHODS: Twenty-seven patients with post-traumatic elbow contracture were followed-up after open arthrolysis for at least 12 months. Before surgery, the mean limitation in extension was 30 and the mean maximum flexion was 83 degrees. A posterior surgical approach was used in 18 patients, and a lateral approach was employed in nine patients. Using the posterior approach, the fibrotic posterior capsule was excised and the ulnar collateral ligament was split. Both the anterior and posterior capsules were released with a lateral approach. RESULTS: The mean flexion increased from 83 degrees to 121 degrees, but the mean extension improved little from -30 degrees to -26 degrees. The mean flexion-extension arc increased from 53 degrees to 95 degrees. According to the elbow evaluation score by the Japanese Orthopaedic Association, both pain and function scales improved significantly. By Hertel's subjective evaluation, the results were good in 13 patients, fair in ten patients, and poor in four patients. Twenty-three patients (85 percent) were satisfied with the results, but four were not satisfied because of residual contracture. These poor results were related to severe soft tissue trauma, residual displacement of intra-articular fragments, and recurrence of heterotopic bone formation. CONCLUSIONS: Tendon lengthening of stiff triceps, accurate reduction of intra-articular fragments, and sharp epiperiosteal resection around the heterotopic bones are essential procedures of open arthrolysis to restore adequate motion in post-traumatic elbow contracture.