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Tetsuo Hotta

Publications and source records attributed to Tetsuo Hotta.

At least 19 recordsLinked to original sources

Margin-free spondylectomy for extended malignant spine tumors: surgical technique and outcome of 13 cases.

STUDY DESIGN: Description of surgical technique and retrospective review of 13 cases. OBJECTIVES: To describe the surgical technique of margin-free spondylectomy and the outcome of 13 cases and to discuss the advantages and limitations of the procedure. SUMMARY OF BACKGROUND DATA: Recently, spondylectomy became a standard procedure by several pioneers. For extended malignant spine tumors involving pedicles or epidural space, however, performing an "en bloc" resection with a tumor-free margin remains a challenge. METHODS: Our procedure consists of a combined anterior and posterior procedure with one or two stages. In the anterior procedure, tumor vertebrae are covered by the pleura or psoas muscles as a barrier. The posterior procedure includes decompression through the intact posterior elements, coverage of the tumor with all possible soft tissue barriers, and en bloc extirpation by rotating the tumor vertebrae around the spinal cord. We performed this procedure in 13 cases: 3 chondrosarcoma, 3 giant cell tumor, 1 osteosarcoma, 1 chordoma, and 5 metastases. RESULTS: Neurologic status and pain improved in all cases except asymptomatic cases. There was no local recurrence, except in 2 cases (chondrosarcoma with extirpation of 5 vertebrae, chordoma with multiple previous surgeries). Two cases of chondrosarcoma were disease-free 14 years and 13 years after surgery, respectively. CONCLUSION: Although the best chance for a cure in extended malignant tumors of the spine is realized through wide resection, the procedure is not yet standardized. Margin-free spondylectomy is technically demanding, but the procedure can be used with a confidence as a more radical surgery for tumors extending to the epidural space and the unilateral pedicle. A key to success is the surgical technique, including a 360 degree dissection around the tumor vertebrae, instrumentation, and removal of the lesion with all possible soft tissues maintained intact to function as a barrier, like the dura mater.

Adolescent↗

Clinical relevance of pathological grades of malignant peripheral nerve sheath tumor: a multi-institution TMTS study of 56 cases in Northern Japan.

BACKGROUND: Malignant peripheral nerve sheath tumor (MPNST) is a relatively rare soft tissue tumor, and its clinical relevance of pathological grades remains obscure. METHODS: Fifty-six cases of MPNST identified from the files of seven oncology centers of the Tohoku Musculoskeletal Tumor Society (TMTS) and National Cancer Center were analyzed for histologic grades, demographics, treatments, and prognostic factors. The average follow-up period was 41 months. RESULTS: Twenty-two men and 34 women with a mean age of 45 years were involved. Forty-four (78.6%) of 56 tumors were in the lower extremity or trunk. Fifty tumors (89%) were classified as high grade, and the remaining six as low grade. Twenty-one (39.6%) of 53 patients who underwent tumor excision developed local recurrences. An axial site and inadequate surgical margin were defined as risk factors for local recurrence. The overall survival rates of the 56 patients were 55.1% at 3 years and 43.3% at 5 years. Univariate analysis of the 56 patients revealed large-sized tumors, metastasis at presentation, and histologically high grade were significantly associated with poor prognosis. Multivariate analysis revealed a large tumor and metastasis at presentation to be independent prognostic factors. CONCLUSIONS: The current study involving 56 patients with MPNST showed the aggressive clinical behavior of the tumor. Large-sized tumors, metastasis at presentation, and high histological grade were related to poor prognosis on univariate analysis, but independency of histological grade was still obscure. In the treatment for a large and high-grade MPNST, an alternative strategy should be further considered.

Adolescent↗

Histological assessment in grafts of highly purified beta-tricalcium phosphate (OSferion) in human bones.

Prominent osteoconductive activity and the biodegradable nature of commercially available beta-tricalcium phosphate (beta-TCP, OSferion) have been documented in animal experiments. We analyzed four cases of involving grafted OSferion in human bone with respect to histological features by routine hematoxylin and eosin staining, silver impregnation, immunohistochemistry and in situ hybridization. OSferion affords early bioresorption by osteoclasts, vascular invasion of macropores and osteoblastic cell attachment on the surface on the ceramic surface 14 days after grafting. Prominent bone formation and direct bone connection between preexisting bone and OSferion were evident 28 days after grafting. Nearly the entire TCP surface was covered by lamellar bone; additionally, active osteoblastic lining and attachment of the osteoclast-like giant cells were not observed 72 weeks after grafting. Silver impregnation revealed the presence of collagen fibrils within probable micropores of OSferion.

Adolescent↗

Clinical outcomes of 54 pelvic osteosarcomas registered by Japanese musculoskeletal oncology group.

BACKGROUND: To determine the clinical outcomes in patients with pelvic osteosarcoma, we reviewed the experience with 54 pelvic osteosarcoma patients. METHODS: Thirty-five patients underwent surgical treatment with 30 undergoing limb salvage procedures and 5 requiring hemipelvectomies. RESULTS: Oncological outcome was continuously disease free in 12 cases, no evidence of disease in 3, alive with disease in 3, died of disease in 34 and died of other causes in 2. The 5-year survival rate was 29.5%. For the patients who underwent surgery, statistically significant prognostic factors were chemotherapy, surgical margin, tumor location in sacrum, efficacy of chemotherapy, local recurrence, metastasis and treatment for metastases. With univariate analysis, local recurrence, surgical margin and tumors resected from the sacrum were significant prognostic factors for metastasis. Surgical margin, tumor resected from sacrum and metastasis were significant prognostic factors for local recurrence. With regard to postoperative functional mobility, patients with tumors resected from the sacrum exhibited poorer functional mobility when compared with other sites. CONCLUSIONS: Patients with tumors located in the sacrum had worse prognosis, and significant prognostic factor for local recurrence and metastasis. Treatment of pelvic osteosarcoma especially located in the sacrum requires new approaches and further improvements in order to ensure successful outcomes.

Adolescent↗

Methylation status of EXT1 and EXT2 promoters and two mutations of EXT2 in chondrosarcoma.

Germline mutation and functional loss of EXT1 or EXT2 are commonly found in multiple osteochondromas and predispose to the development of chondrosarcoma. Mutations of EXT1 and EXT2 have rarely been detected in sporadic secondary chondrosarcomas from osteochondroma; these frequently display loss of heterozygosity at the EXT1 and EXT2 loci, but primary chondrosarcomas typically do not. To evaluate promoter methylation (which is an epigenetic gene silencing mechanism) of EXT1 and EXT2, we performed methylation-specific polymerase chain reaction (PCR) for 20 chondrosarcoma cases (12 primary, 3 secondary to osteochondroma, 2 secondary to enchondromatosis, 2 extraskeletal ordinary, and 1 clear cell) and in five cell lines. In addition, mutation analysis of the EXT1 and EXT2 coding regions was performed using PCR-single-strand conformation polymorphism and sequencing analysis for 12 of the 20 chondrosarcoma cases (8 primary, 1 secondary to enchondromatosis, 1 secondary to osteochondroma, and 2 extraskeletal ordinary) and five cell lines. Promoter methylation of EXT1 and EXT2 was not detected in any of the cases, and both EXT1 and EXT2 were expressed in all cell lines. Two missense mutations in EXT2 (D227E and R299H) were detected among the chondrosarcoma cases. When considering tumor development in primary chondrosarcoma, we should include mutations in EXT2, along with the status of other members of the EXT gene family.

5' Untranslated Regions↗

Comparison of hydroxyapatite and beta tricalcium phosphate as bone substitutes after excision of bone tumors.

Long-term results are reported in 23 patients and short-term results in 30 patients presenting with bone tumors treated by curettage or resection followed by implantation of hydroxyapatite (HA) or highly purified beta-tricalcium phosphate (beta-TCP), respectively. Mean follow-up was 97 and 26 months in cases involving HA implantation and beta-TCP implantation, respectively. Radiographs revealed HA incorporation into host bone in all but two cases; moreover, no obvious evidence of HA biodegradation was observed. A single patient exhibited late deformity following implantation of HA. All grafted beta-TCP was, at least partially, absorbed and replaced by newly formed bone. The mean period required for the disappearance of radiolucent zones between the ceramics and host bone was 17 weeks in HA and 9.7 weeks in beta-TCP. Highly purified beta-TCP appears to be advantageous relative to HA for surgical intervention in bone tumors consequent to the nature of remodeling and superior osteoconductivity.

Adolescent↗

Ischial weight-bearing brace after the infection of megaprosthesis a salvage method for resection arthroplasty.

We report a case of successful control of the infection of megaprosthesis in the distal femur. Deep infection occurred 5 years after the resection of the osteosarcoma and reconstruction with cementless megaprosthesis. Multiple debridement and intravenous administration of antibiotics did not eradicate the infection. He developed aortitis syndrome (Takayasu's arteritis) and needed to undergo steroid therapy. Total removal of the prosthesis achieved good control of the infection, and the patient had started prednisolone therapy. The patient can walk alone with an ischial weight-bearing brace. Removal of the endoprosthesis and application of ischial weight-bearing brace may be a possible option for the treatment of infected megaprosthesis.

Adult↗

Establishment and characterization of a novel myxofibrosarcoma cell line.

We established a novel human myxofibrosarcoma cell line NMFH-1 and analyzed it with spectral karyotyping and comparative genomic hybridization (CGH). NMFH-1 cells are composed of two different types of cells, small, spindle-shaped mononuclear cells and bizarre multinucleated giant cells, which were maintained in vitro over 200 passages. Xenografted tumor showed typical features of myxofibrosarcoma, which included bizarre multinucleated giant cells. Cytogenetic analyses revealed complex abnormalities, including a t(17;22)(q2?2;q13), which has been found in dermatofibrosarcoma protuberans. Subsequent reverse-transcription polymerase chain reaction revealed that the cell line did not have the COL1A1-PDGFB gene fusion. Significant gains of the 1q12 approximately q23 and 8q13 approximately qter regions and loss of the 9p21 approximately pter and 13q12 regions often found in MFH were observed by CGH analysis. We investigated the origin of multinucleated giant cells in xenografted tumor through DNA in situ hybridization. In this system, the human-specific Alu sequence and the mouse L1 sequence were used as specific cell markers of identity. In situ hybridization revealed neoplastic proliferation of the multinucleated giant cells of human origin.

Aged↗

Sclerosing epithelioid fibrosarcoma with der(10)t(10;17)(p11;q11).

Sclerosing epithelioid fibrosarcoma is a recently described, rare mesenchymal neoplasm. We report a case of sclerosing epithelioid fibrosarcoma that occurred in the lower leg of a 48-year-old man. The karyotype of the tumor exhibited der(1)t(1;10)(p31;p11), der(10)t(10;17)(p11;q11), and der(17) t(11;17)(?;q11). Rearrangement of 10p11 was also found in one previous reported case of this uncommon tumor.

Chromosome Aberrations↗

Successful surgical treatment of angiosarcoma of the spine: a case report.

STUDY DESIGN: Case report. OBJECTIVES: To report primary angiosarcoma of the T8 vertebra, which was successfully managed with en bloc spondylectomy and postoperative chemotherapy. SUMMARY OF BACKGROUND DATA: To the best of our knowledge, the present case is the first documented example of successful treatment of angiosarcoma of the spine. METHODS: Angiosarcoma of the eighth thoracic vertebra was diagnosed in a 48-year-old man with impending neurologic deficit. Imaging findings revealed a nonspecific high-grade lesion. A total spondylectomy of T8 by en bloc resection was performed. The defect of the vertebral body was reconstructed with a apatite-wollastonite glass ceramic prosthesis; moreover, the T6-T10 vertebrae were instrumented by the pedicle screw, hook and rod system. The histologic diagnosis of the excised specimen was high-grade angiosarcoma. Postoperative chemotherapy was implemented to prevent local recurrence and distant metastasis. RESULTS: No sign of local recurrence or metastasis was evident 5 years after surgery. CONCLUSION: This case is the first documented example of successful treatment of angiosarcoma of the thoracic spine. Radiologic findings were nonspecific; consequently, correct diagnosis was established by pathologic examination. Immediate, aggressive operative treatment and postoperative adjuvant chemotherapy afforded a satisfactory outcome.

Antineoplastic Combined Chemotherapy Protocols↗

High-level expression of the coxsackievirus and adenovirus receptor messenger RNA in osteosarcoma, Ewing's sarcoma, and benign neurogenic tumors among musculoskeletal tumors.

PURPOSE: The sensitivity of human tumor tissues to infection with recombinant adenoviruses correlates with the expression of the coxsackievirus and adenovirus receptor (CAR). CAR has been shown to function as the primary receptor for adenoviruses and to play a critical role in adenovirus entry into host cells. It is important for clinical gene therapy to determine the expression level of CAR in tumor tissues. EXPERIMENTAL DESIGN: We analyzed the expression of CAR mRNA in 154 musculoskeletal tumor tissues from 154 patients and 10 normal mesenchymal tissues from 3 patients using reverse transcription-PCR and real-time quantitative PCR. An adenovirus infection assay was performed in two cell lines that were established from CAR-positive osteosarcoma tissue and CAR-negative malignant fibrous histiocytoma tissue. RESULTS: Ninety-nine of 154 tumors were detected as CAR positive by reverse transcription-PCR. We found that the expression levels of CAR mRNA varied markedly between different tumors as determined by real-time quantitative PCR. CAR mRNA was expressed at high levels in osteosarcoma, Ewing's sarcoma, neurofibroma, and schwannoma; at intermediate levels in exostosis, giant cell tumor, liposarcoma, synovial sarcoma, malignant peripheral nerve sheath tumor, and hemangioma; and at low levels in alveolar soft part sarcoma and desmoid. Whereas the osteosarcoma cell line that expressed a high level of CAR mRNA, like its parent tumor, had a high efficiency of adenovirus infection, the malignant fibrous histiocytoma cell line with almost undetectable expression of CAR mRNA, like its parent tumor, had a low efficiency of infection. CONCLUSIONS: Our data showed the great variations in CAR mRNA expression among human musculoskeletal tumors and mesenchymal tissues and implicated the potential usefulness of adenoviral vectors in gene therapy for osteosarcoma, Ewing's sarcoma, neurofibroma, and schwannoma. Efficient transduction with adenovirus for gene therapy could be realized in appropriate, sensitive tumor types.

Adenoviridae↗

CA125 expression in epithelioid sarcoma.

OBJECTIVE: There has been no report on useful immunohistological markers for epithelioid sarcoma (ES) so far. The purpose of this study is to evaluate the positivity and specificity of CA125 as a marker for the correct diagnosis of ES. METHODS: This study was performed in 11 patients with ES (nine men and two women; distal type: 10 cases; proximal type: one case), 78 patients with other soft tissue tumors and nine with benign granulomas. The other soft tissue tumors consisted of six synovial sarcomas, six clear cell sarcomas, eight leiomyosarcomas, six rhabdomyosarcomas, five malignant peripheral nerve sheath tumors, ten malignant fibrous histiocytomas, 17 desmoid tumors, 14 liposarcomas, six squamous cell carcinomas (cutaneous SCC of the distal extremities), two rheumatoid nodules and seven foreign body granulomas. Immunohistochemical analysis for CA125 was performed for these 89 soft tissue tumors and nine granulomas using a labeled streptavidin biotin method. Immunohistochemical analysis of epithelial membrane antigen, cytokeratin, carcinoembrionic antigen, vimentin and CD34 was performed only for the 11 ES patients. RESULTS: CA125 was strongly expressed in 10 out of the 11 ES patients. EMA, cytokeratin and vimentin were also positive in all the cases. CEA was positive in two of the 11 patients. Immunohistochemical study in six ES patients showed expression of CD 34. The other 78 soft tissue tumors and nine granulomas did not express CA125. CONCLUSION: This study clearly revealed the specificity and positivity of CA125 in ES. These data indicate that CA125 may be a useful tumor marker for diagnosing ES.

Adolescent↗

Diagnosis of peripheral nerve sheath tumors around the pelvis.

OBJECTIVE: To distinguish between benign and malignant peripheral nerve sheath tumors around the pelvis. METHODS: A retrospective study of 30 patients with benign and malignant peripheral nerve sheath tumors located around the pelvis was performed. Clinical, imaging and histological features of 19 benign and 11 malignant peripheral nerve sheath tumors around the pelvis were reviewed retrospectively. RESULTS: Nearly all patients exhibited pain at presentation in cases involving both benign and malignant tumors. Although tumor size, duration of symptoms and presence of sensory disturbance possessed little value in differential diagnosis, severe motor weakness was observed exclusively in patients presenting with malignant tumors. On CT or MRI, central enhancement was apparent in 11 of the 19 benign tumors; in contrast, central enhancement was evident in one of the 11 malignant tumors. Fine needle aspiration cytology was performed in 11 tumors; correct diagnosis was achieved in four tumors. Core needle biopsy was performed in five tumors, all of which were correctly diagnosed with no neurological deficits. Immunohistochemically, all benign tumors were diffusely positive for S-100 protein, whereas malignant tumors were negative or focally positive for S-100 protein. Ki-67 index was less than 4% in all benign tumors; additionally, this index was 7-36% in malignant tumors. CONCLUSION: Central enhancement pattern on imaging studies strongly suggests a benign tumor; in contrast, severe motor weakness suggests malignant lesions. Core needle biopsy was reliable with respect to preoperative diagnosis.

Adolescent↗

Plexiform schwannoma of the forearm.

We report a case of plexiform schwannoma located in the flexor muscles of the forearm in the absence of other signs of neurofibromatosis or schwannomatosis. Magnetic resonance examination revealed a multinodular irregular inhomogeneous mass. Some nodules displayed a peripheral, high intensity rim and a central low intensity (target sign) on T2-weighted images. Pre-operative diagnosis of the rare plexiform schwannoma may be possible with careful imaging examination for the target sign.

Adult↗

Presacral multiple cellular schwannomas.

STUDY DESIGN: Case report. The authors documented a patient presenting with multiple cellular schwannomas along the sciatic nerve. The patient had no stigmas of neurofibromatosis. Thirteen tumors were resected via both anterior and posterior approaches. OBJECTIVES: To point out that presacral cellular schwannoma can display multiple occurrences. SUMMARY OF BACKGROUND DATA: Cellular schwannoma, which is a well-recognized variant of benign schwannoma, is often misdiagnosed as sarcoma. The tumor is usually a solitary lesion. MATERIALS AND METHODS: Medical history, physical findings, imaging features, and histologic findings were reviewed in a case of multiple cellular schwannomas in the presacral regions. RESULTS: A 39-year-old woman without any evidence of neurofibromatosis underwent removal of presacral multiple tumors along the sacral nerve root. Nineteen years after the surgery, the tumors recurred with multiple fashion. Thirteen tumors were resected via both anterior and posterior approaches. Immunohistochemical analysis of S-100 protein and Ki-67 were beneficial with respect to differentiation of cellular schwannoma from malignant peripheral nerve sheath tumor. CONCLUSIONS: The present case illustrated that multiple cellular schwannomas can develop in nonneurofibromatosis patients.

Adult↗