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Biomedical subjects

Masami Hosaka

Publications and source records attributed to Masami Hosaka.

16 recordsLinked to original sources
PubMed2006

Osteosarcoma arising from giant cell tumor of bone ten years after primary surgery: a case report and review of the literature.

Giant cell tumor of the bone (GCT) is a relatively uncommon tumor. It is characterized by the presence of multinucleated giant cells. GCT is a primary benign tumor but may evolve into a malignant tumor, usually after irradiation. We report a rare case of osteosarcoma arising ten years after the primary surgery for GCT without radiation. A 45-year-old woman presented with severe right knee pain after suffering contusion. Roentgenogram revealed a bone tumor in the lateral femoral condyle of the right knee. Histopathological examination demonstrated the features of GCT, and treatment consisted of curettage and bone grafting. Four months after the operation, multiple lung metastases of GCT occurred, which were treated by partial lobectomy. Ten years after the primary treatment, severe knee pain recurred. Roentgenogram and magnetic resonance imaging (MRI) demonstrated a destructive lesion in the lateral condyle of the right knee. Histopathological examination demonstrated a lacy pattern of osteoids and abnormal mitoses in the aggregated atypical mononuclear cells, indicating osteosarcoma. Despite above-knee amputation and chemotherapy, scapular and lung metastases developed and the patient died five months after above knee amputation. One may question whether the primary GCT contained some malignant cells. However, given the aggressiveness of the malignant tumor, this is unlikely. The recurrence of pain and aggravation of bone destruction many years after the primary treatment suggest malignant transformation of GCT.

Bone Neoplasms↗
PubMed2006

Diffuse large B-cell lymphoma arising in a patient with neurofibromatosis type I and in a patient with neurofibromatosis type II.

We experienced two unusual cases of diffuse large B-cell lymphoma (DLBCL), which occurred in a patient with neurofibromatosis type I and a patient with neurofibromatosis type II. B-cell lymphoma is one of the most common phenotypic subgroups of malignant lymphoma. Neurofibromatosis I is characterized by café-au-lait spots and neurofibromas. Neurofibromatosis II typically consists of bilateral schwannomas of the acoustic nerve. Malignant lymphomas rarely coexist with neurofibromatosis I, and no coexistence with neurofibromatosis II has been reported. The patient with neurofibromatosis I was a 50-year-old Japanese woman, clinically manifesting von Recklinghausen's disease since infancy, who noticed an egg-sized tumor in her shoulder. The patient with neurofibromatosis II was a 39-year-old Japanese man who noticed multiple soft tissue tumors in his neck, buttock, and elbow. Biopsied materials from both cases were examined by multiparameter methods, including flow cytometry. Flow cytometry revealed large-scale cells to be tumor cells, and they were positive for CD19, CD20, and CD22. Both patients received chemotherapy, and the tumors disappeared. The patient with neurofibromatosis I was alive without recurrence at 4 years after treatment, while the patient with neurofibromatosis II died of recurrence. To the best of our knowledge, this is the first case of malignant lymphoma arising in a neurofibromatosis II patient. As for neurofibromatosis I, there were some reports about occurrence of malignant lymphoma. It is important to be aware of possibility of association of malignant tumors not only of the nervous system but also of unrelated to the nervous system when tumors appear in neurofibromatosis patients.

Adult↗
PubMed2006

Osteosarcoma in a patient with neurofibromatosis type 1: a case report and review of the literature.

Neurofibromatosis type 1 (NF1) or von Recklinghausen's disease is a genetic disease generally characterized by café-au-lait spots and neurofibromas. Malignant tumors of the nervous system, such as malignant schwannomas, gliomas, or astrocytomas, have been well known to coexist with neurofibromatosis. However, occurrence of malignant tumors unrelated to the nervous system is rare. We report an unusual case of a 29-year-old NF1 female suffering from malignant peripheral nerve sheath tumor (MPNST) that eventually developed osteosarcoma in the proximal femur. Osteosarcoma is the most common high-grade malignant bone tumor in which the neoplastic cells produce osteoid. At 23 and 24 years old, she underwent excision of MPNST in the left posterior thigh. No osteosarcomatous portion was identified in these specimens. The patient underwent postoperative chemotherapy. At 29, left proximal thigh pain and swelling appeared. Computed tomography demonstrated cortical bone destruction in the left proximal femur where MPNST occurred. Magnetic resonance imaging revealed extraskeletal growth of the tumor. Bone scintigraphy demonstrated increased uptake in the left proximal femur. Hip disarticulation was performed. The removed tumor was composed of highly anaplastic cells. Lace-like irregular osteoid formation was observed among the tumor cells. MPNST component was totally absent. The tumor was diagnosed as osteoblastic type osteosarcoma. Two months after disarticulation the patient died of bilateral pulmonary metastasis. The correlation between the histogenesis of osteosarcoma and the genetic abnormality in NF1 patients has not been elucidated, but the finding of osteosarcomatous transformation in this case suggests the divergent cellular differentiation to mesenchymal malignant tumors of neuroectodermal tissue in NF1 patients.

Adult↗
PubMed2006

A classic adamantinoma arising from osteofibrous dysplasia-like adamantinoma in the lower leg: a case report and review of the literature.

Adamantinoma is known as a low-grade malignant biphasic tumor. Classic adamantinoma is in general characterized by admixture of both epithelial and osteofibrous components that are associated with various proportions and differentiation patterns. Osteofibrous dysplasia (OFD) is a self-limited benign fibro-osseous lesion of bone during infancy and childhood. OFD-like adamantinoma is characterized by predominance of osteofibrous tissues, in which small groups of epithelial cells are only detected by careful search or immunohistochemistry. There have been controversies as to the potential correlation among OFD, OFD-like adamantinoma and classic adamantinoma. We report an unusual case of adamantinoma arising in the tibia, with an extensive review of the literature. The present findings suggest a direct correlation between OFD-like adamantinoma and classic adamantinoma. At the age of 12 years, the tibial biopsy lesion was diagnosed as OFD. At the age of 23, the lesion became larger and more destructive on x-ray films. The biopsy lesion was diagnosed as classic adamantinoma. Wide excision was performed. The primary lesion was retrospectively diagnosed as OFD-like adamantinoma because of presence of keratin-positive epithelial cells within the stroma. At five years after surgery, the patient was free from recurrence or metastasis. The retrospective histological findings of OFD-like adamantinoma in the original biopsy and of a classic adamantinoma in all sections of the later resection specimen raised the possibility of an unusual progression of OFD-like adamantinoma to a classic adamantinoma. The present case and the literature review suggest that an OFD-like adamantinoma may be a precursor lesion of classic adamantinoma. Therefore, the possibility of progression of OFD-like adamantinoma to a classic adamantinoma should be kept in mind, particularly when the destructive changes are seen radiologically.

Adamantinoma↗
PubMed2006

Establishment and characterization of a clonal human extraskeletal Ewing's sarcoma cell line, EES1.

Ewing's sarcoma, a small round cell sarcoma arising in soft tissue as well as the bone, is one of the most malignant tumors in children and young adults. Few established cell lines of extraskeletal Ewing's sarcoma (EES) have been reported, which made it difficult to examine the biological features of EES. Therefore, we have established a new clonal cell line of EES. We report its morphological characters, results of chromosomal and immunohistochemical analysis. A piece of tumor obtained from the 18-year-old female patient with EES was xenografted in a nude mouse. In vitro subcultured cells were then obtained from this xenograft. A clonal cell line was subsequently established by limiting dilution and designated EES1. EES1 cells had a doubling time of 24 hours. In the xenografted tumor, the cells expressed vimentin, CD99 (MIC2), neuron specific enolase (NSE) and cytokeratin. The original tumor cells also expressed vimentin, CD 99, and NSE, but was negative for cytokeratin. The morphological and immunohistochemical features of this cell line established, except for cytokeratin expression, were consistent with those of the primary tumor. Cytogenetic analysis of EES1 revealed chromosomal translocation of t(11; 12)(q24;ql2). The chimeric fusion of the Ewing's sarcoma gene in band 22q12 with the Friend leukemia virus integration-1 gene in band 11q24 was also demonstrated. Fluorescence in situ hybridization further confirmed the presence of translocation involving the Ewing's sarcoma gene in both the primary tumor and EES1 cells. In conclusion, we have established a human EES cell line EES1, which will provide a useful model for studying various aspects of human EES.

Adolescent↗
PubMed2006

Recurrent Crow-Fukase syndrome associated with increased serum levels of vascular endothelial growth factor: a case report and review of the literature.

Crow-Fukase syndrome (CFS) is a rare multi-system disorder, characterized by polyneuropathy, organomegaly, endocrinopathy, M-proteins, skin changes and anasarca, with or without myeloma. The pathophysiology, diagnosis, and treatment of CFS are controversial. CFS may be associated with the overproduction of vascular endothelial growth factor (VEGF). However, there have been no reports of monitoring the serum VEGF level after recurrence, to the best of our knowledge. We report a 54-year-old man with CFS presenting with a 3-year history of ascites, anasarca, weakness of the lower extremities, and plasmacytoma in the scapula. At the initial examination, the VEGF level was 1,590 pg/ml (the VEGF level of a healthy control, 78.4 +/- 75.2 pg/ml). After initial treatment with chemotherapy and irradiation of the affected shoulder, the VEGF level decreased to 154 pg/ml and the symptoms disappeared. Twenty one months later, gate disturbance and anasarca recurred, and the VEGF level was over 2,000 pg/dl. After total scaplectomy, the VEGF level decreased to 730 pg/dl and the symptoms disappeared. The serum level of VEGF well correlated to the clinical course of the patient. In conclusion, measurement of the VEGF level is useful for diagnosing CFS and for monitoring its clinical course.

Bone and Bones↗
PubMed2006

Operative treatment by external fixation for polyostotic fibrous dysplasia in the elbow joint. A case report.

Fibrous dysplasia is a condition in which normal skeletal bone changes into fibroblastic stroma and immature bone. We report our experience of a unilateral external fixator applied for the treatment of polyostotic fibrous dysplasia arising in the elbow. The patient was a 38 year-old man. His main complaint was left elbow pain and a mass, which increased in size. The histological diagnosis from the needle biopsy was fibrous dysplasia. An articulated unilateral external fixator was used for immobilization. The lesions in the left distal humerus and proximal ulna were curetted and cancellous bone was packed into the cavity. An external fixator (Elbow Fixator, Orthofix Inc., McKinney, USA) was applied from the proximal humeral shaft to the distal ulnar shaft for postoperative tentative immobilization and preservation of the affected elbow joint movement. At two years after the operation, the range of motion of the affected elbow was 90 degrees in flexion and -35 degrees in extension. The range of motion in pronation and supination of the left forearm was normal. No evidence of recurrence was noted at two years after surgery. The merits of using an external fixator were: an earlier start of range of motion exercises, and a decrease in the risk of tumor dissemination.

Adult↗
PubMed2005

Desmoplastic melanoma arising in the ankle.

Desmoplastic melanoma (DM) is an uncommon but potentially devastating malignancy that can be cured with early recognition and surgery. DM often occurs in elderly men and develops on sun-exposed areas with the head and neck being the most common site of origin followed by the extremities and trunk. We report a rare case of DM occurring in the ankle in an 85-year-old woman. Magnetic resonance imaging of the affected ankle showed a 4.3 x 4.3 x 2.5 cm sized mass which was located in the subcutaneous region, attached to lower leg muscles, the fibula, and the calcaneus. No metastasis to the lung, liver, bones or abdominal lymph nodes was found. In spite of below knee amputation, the patient died of lung metastases 10 months after surgery. Among several factors causing early death of the patient, a large size, a deep location and rapid growth of the tumor seem to be most important.

Aged, 80 and over↗
PubMed2005

Lipofibromatosis arising in a pediatric forearm--a case report.

Lipofibromatosis is a rare pediatric tumour described by Fetsch et al. in 2000. There have been few reports about this tumour and few descriptions of its imaging features. We report a case of this tumour forming a slowly growing, painless mass arising in the forearm of a ten-month-old boy. This is the first report of lipofibromatosis to present the radiological findings before surgery. Plain radiography and computed tomography demonstrated a tumour located just beneath the fascia. Ultrasonic study showed a hyperechoic tumour of 33 x 30 x 7 mm in size. Magnetic resonance imaging demonstrated a multilobular mass with high signal intensities on both T1/T2 weighted images. Macroscopically, the tumour was poorly marginated. Microscopically, the tumour was composed of abundant adipose tissues transversed by fibroblastic bands. The adipose tissue occupied over 70% of the tumour. Adipocytes and fibroblastic element infiltrated into the normal skeletal muscle tissues. Recognition of this clinical entity is needed, especially in cases of lipomatous tumours arising in pediatric hands and feet.

Fibroma↗
PubMed2005

Spindle cell lipoma showing biphasic pattern--a case report.

This is a case report of a rare spindle cell lipoma (SCL) showing an unusual biphasic pattern with radiological findings and histological features. SCL is most common in men between the ages of 45 and 65 and it is reported that the tumor has a tendency to occur in the posterior neck area. This case was a 75-year old man with a 7 x 5 cm subcutaneous tumor in the posterior neck. Magnetic resonance imaging (MRI) examination demonstrated an unusual biphasic pattern. The signal intensities on T1 and T2 weighted images of the outer layer of the mass were high, and were suppressed on fat suppression views and showed no enhancement after gadolinium (Gd) injection. There was a low signal intensity area of 15 mm in diameter on both T1 and T2 weighted images, in the central part of the mass. This area was well enhanced with the Gd compound. Also macroscopically it could be seen that the excised tumor consisted of two different tissue components. Microscopically, the outer yellowish portion was mainly composed of mature adipose tissue. The inner whitish fibrous portion contained a mixture of small and uniform spindle-shaped cells, collagen fibers, and scattered mature adipose cells. No malignancy was observed. The diagnosis of spindle cell lipoma was made. No recurrence was seen seven months after marginal excision.

Aged↗
PubMed2004

Giant cell formation through fusion of cells derived from a human giant cell tumor of tendon sheath.

Although the mechanism of multinucleation in giant cell tumors of tendon sheath (GCTTS) remains unknown, two mechanisms have been proposed: one is cell fusion and the other amitotic division. The purpose of this study was to clarify the multinucleation process of cultured cells from GCTTS using an in vitro fluorescent cell membrane labeling technique. Cultured GCTTS cells obtained from a 7-year-old Japanese girl were divided into two groups, one for PKH-2 (green) staining and the other for PKH-26 (red) staining. After staining with the dyes, the cell populations were mixed and observed with a fluorescent microscope on the 4th and 14th days after mixing. On both the 4th and 14th days, the cultured GCTTS giant cells showed a mosaic of green and red colors, thus indicating cell membrane fusion. Images of double fluorescent labeled giant cells indicated cell fusion of mononucleated stromal cells that lead to multinucleated giant cells in these GCTTS cell cultures. These findings suggest that multinucleation in GCTTS results from the fusion of mononuclear stromal cells in vitro.

Animals↗
PubMed2004

A two- stage surgery for severe femoral neck deformity due to fibrous dysplasia: a case report.

Various kinds of surgical treatments have been reported for varus deformity of the proximal femur due to fibrous dysplasia. We report a case of two-stage corrective osteotomy for severe varus-retroversion deformity of the femoral neck due to monostotic fibrous dysplasia. The patient was an 18 year-old man. On initial examination, the spina malleollar distance was 88 cm on the right side and 83 cm on the left. Plain radiography showed prominent varus deformity of his left proximal femur. The morphology was 130 degrees on the right side and 85 degrees on the left. Computed tomography revealed 60 degree retroversion of the femoral neck. A two-stage surgery was performed, consisting of curettage and bone grafting followed by corrective osteotomy 16 months later. A 55 degree valgus osteotomy was performed in the subtrochanteric region. After osteotomy and 40 degree internal rotation of the shaft, a 130 degree angle plate was used for osteosynthesis. Postoperative radiological examination showed a morphology of 140 degrees and computed tomography revealed a 20 degree retroversion of the femoral neck. No recurrence or varus deformity was seen at four years after surgery. Although the leg length discrepancy was 2.5 cm, the patient had no difficulty in one foot standing and no restriction of ADL (activity of daily living). The well-known progressive varus Shepherd's crook deformity in the polyostotic form of fibrous dysphasia is associated with limb shortening, limping, and occasionally chronic fatigue fractures with disabling pain. Various kinds of surgical treatments have been reported for this type of varus deformity. Curettage and bone grafting is one of the most common and simple treatments. However, this method often gives bad results as the grafted bones are absorbed and that the progress of varus deformity van not to be prevented. We report a case of two-stage corrective osteomy forsevere varus-retroversion deformity of the femoral neck due to monostotic fibrous dysplasia.

Activities of Daily Living↗
PubMed2003

Pigmented villonodular synovitis of the knee with lesions of the bursae.

Five patients with pigmented villonodular synovitis of the knee associated with lesions of the posterior periarticular bursae were treated by posterior exploration, excision of the bursal tissue, and routine anterior synovectomy of the knee. It was hypothesized that complete excision of the posterior periarticular bursae would reduce the high recurrence rate of pigmented villonodular synovitis of the knee. To completely excise the periarticular lesions, two posterior oblique skin incisions were used. In all patients, periarticular lesions were seen clearly on computed tomography scans after air arthrography and on magnetic resonance imaging scans. The patients were followed up for 6.9 years on average (range, 3.3-8.1 years) after surgery. One of five patients required reoperation because of recurrence. Two of five patients required manual mobilization after release of the intraarticular adhesion. However, in the remaining three patients, who started continuous passive motion exercise immediately after surgery, full range of motion was restored within 3 months after the operation. Complete excision of the periarticular lesions is recommended to reduce the high recurrence rate, and continuous passive motion exercise immediately after surgery also is recommended to prevent reduction of range of motion.

Adult↗
PubMed2003

Synovial sarcoma with massive ossification--a case report.

Synovial sarcoma with extensive osteoid production is rare. We report a case of synovial sarcoma of monophasic type with massive ossification. The diagnosis was confirmed by reverse-transcripitase polymerase chain reaction (RT-PCR). The patient was an-81-year-old woman with recurrent synovial sarcoma in her right knee. The tumor was primarily excised in 1989. It recurred and was removed again in 1996. However, in 1999 a painful mass appeared in the same site. Preoperative plain radiography and computed tomography revealed a 5 x 5 cm soft tissue mass with extensive ossification in the medial side of the right knee joint. The tumor was widely excised in 2000. Soft X-ray examination revealed a trabecular pattern of ossification in the excised tumor. Microscopically the tumor was composed of hypercellular spindle cells with fascicular arrangement with prominent ossification but no epithelial component. The tumor cells were positive for vimentin and focally positive for cytokeratin. The tumor expressed a sequence of SYT-SSX1 fusion gene transcript demonstrated by RT-PCR. Twelve years long survival of the present case without metastasis in spite of repeated recurrence suggests a better prognosis of synovial sarcomas with ossification.

Aged↗
PubMed2002

Pycnodysostosis associated with spondylolysis.

We report 23 years of observation of a patient with pycnodysostosis associated with progressive spondylolysis. There have been very few papers describing the development of spondylolysis associated with pycnodysostosis as confirmed by serial X-ray examinations. The diagnosis was made by the patient's typical bird-like face, plain radiological findings, and repeated tibial fractures. At the age of 8 years, spondylolysis of L4 was observed. The spondylolysis had increased in number to 4 by the age of 24 years, that is, L2 through L5.

Adult↗