記述言語：英語   掲載種別：研究論文（学術雑誌）  

BACKGROUND: Synovial sarcoma is a rare malignant soft-tissue tumor that is prevalent in adolescents and young adults, and poor prognosis has been reported in patients with metastatic lesions. Chimeric antigen receptor (CAR) T-cell therapy is an emerging novel therapy for solid tumors; however, its application in synovial sarcoma has not yet been explored. METHODS: A novel human epidermal growth factor receptor 2 (HER2)-targeted CAR containing scFv-FRP5, CD8α hinge and transmembrane domains as well as 4-1BB costimulatory and CD3ζ signaling domains was developed. Three synovial sarcoma cell lines that expressed the fusion transcript SS18-SSX1/2/4 were used in the study. Cytokine secretion assay, cytotoxicity assay, and real-time cell analysis experiments were conducted to confirm the function of T cells transduced with the CAR gene. RESULTS: High cell-surface expression of HER2 was observed in all the cell lines. HER2-targeted/4-1BB-costimulated CAR T cells specifically recognized the synovial sarcoma cells, secreted interferon gamma and tumor necrosis factor alpha, and exerted cytotoxic effects in these cells. CONCLUSION: To the best of our knowledge, this is the first study to indicate that HER2-targeted CAR T cells are directly effective against molecularly defined synovial sarcoma cells. Furthermore, our findings might set the basis for developing improved CAR T cell-based therapies for chemo-refractory or relapsed synovial sarcoma.

DOI： 10.1016/j.tranon.2021.101227

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記述言語：日本語   出版者・発行元：(株)北隆館  

骨軟部腫瘍における免疫環境の研究は未開拓分野である。筆者らは肉腫の一部に自然寛解がみられることより、骨軟部腫瘍においても免疫環境が予後に影響を持つものと考え研究を続けている。これまでHLA class Iの腫瘍細胞おける発現、CD8陽性リンパ球、CD163陽性マクロファージ浸潤を解析してきたが、組織型ごとに特徴や予後に関する影響も異なっていた。今後はCAR-T細胞を用いた細胞療法等が新たな治療戦略になることを期待し研究を続けている。(著者抄録)

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：英語   掲載種別：研究論文（学術雑誌）  

The characteristics of the tumor immune microenvironment remains unclear in liposarcomas, and here we aimed to determine the prognostic impact of the tumor immune microenvironment across separate liposarcomas subtypes. A total of 70 liposarcoma patients with three subtypes: myxoid liposarcoma (n = 45), dedifferentiated liposarcoma (n = 17), and pleomorphic liposarcoma (n = 8) were enrolled. The presence of tumor infiltrating lymphocytes (CD4+ , CD8+ , FOXP3+ lymphocytes) and CD163+ macrophages and expression of HLA class I and PD-L1 were assessed by immunohistochemistry in the diagnostic samples; overall survival and progression-free survival were estimated from outcome data. For infiltrating lymphocytes and macrophages, dedifferentiated liposarcoma and pleomorphic liposarcoma patients had a significantly higher number than myxoid liposarcoma patients. While myxoid liposarcoma patients with a high number of macrophages were associated with worse overall and progression-free survival, dedifferentiated liposarcoma patients with high macrophage numbers showed a trend toward favorable prognosis. Expression of HLA class I was negative in 35 of 45 (77.8%) myxoid liposarcoma tumors, whereas all dedifferentiated liposarcoma and pleomorphic liposarcoma tumors expressed HLA class I. The subset of myxoid liposarcoma patients with high HLA class I expression had significantly poor overall and progression-free survival, while dedifferentiated liposarcoma patients with high HLA class I expression tended to have favorable outcomes. Only four of 17 (23.5%) dedifferentiated liposarcomas, two of eight (25%) pleomorphic liposarcomas, and no myxoid liposarcoma tumors expressed PD-L1. Our results demonstrate the unique immune microenvironment of myxoid liposarcomas compared to other subtypes of liposarcomas, suggesting that the approach for immunotherapy in liposarcomas should be based on subtype.

DOI： 10.1007/s00262-021-02928-1

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(一社)日本脊椎脊髄病学会  

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記述言語：日本語   出版者・発行元：(一社)日本脊椎脊髄病学会  

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記述言語：日本語   出版者・発行元：(一社)日本脊椎脊髄病学会  

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記述言語：日本語   出版者・発行元：(一社)日本脊椎脊髄病学会  

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記述言語：日本語   出版者・発行元：(一社)日本脊椎脊髄病学会  

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記述言語：日本語   出版者・発行元：(一社)日本脊椎脊髄病学会  

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記述言語：日本語   出版者・発行元：(一社)日本脊椎脊髄病学会  

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記述言語：日本語   出版者・発行元：(一社)日本脊椎脊髄病学会  

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記述言語：日本語   出版者・発行元：(一社)日本脊椎脊髄病学会  

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記述言語：日本語   出版者・発行元：(一社)日本脊椎脊髄病学会  

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記述言語：日本語   出版者・発行元：(一社)日本脊椎脊髄病学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：英語   掲載種別：研究論文（学術雑誌）  

A 21-year-old man consulted our hospital for treatment of a spinal giant cell tumor (GCT) of Enneking stage III. Lower lumbar-spine tumors and severe spinal canal stenosis are associated with high risk for surgical mor-bidity. Stability was temporarily secured with a percutaneous pedicle screw fixation in combination with deno-sumab, which shrank the tumor. Total en bloc spondylectomy was then performed 6 months after initiation of denosumab, and the patient was followed for 3 years. There was no local recurrence, and bony fusion was obtained. Minimally invasive surgery and denosumab allowed safer and easier treatment of a collapsing lower lumbar extra-compartmental GCT.

DOI： 10.18926/AMO/61442

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記述言語：日本語   出版者・発行元：新潟整形外科研究会  

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記述言語：日本語   出版者・発行元：新潟整形外科研究会  

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記述言語：日本語   出版者・発行元：新潟整形外科研究会  

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記述言語：日本語   出版者・発行元：新潟整形外科研究会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：英語   掲載種別：研究論文（学術雑誌）  

BACKGROUND AND AIM: The pathophysiology of rheumatoid arthritis (RA) is characterized by excess production of pro-inflammatory cytokines, including tumor necrosis factor-α (TNF-α), interleukin-1β (IL-1β), and interleukin-6 (IL-6) by neutrophils and macrophages in synovium. Additionally, these cytokines promote the production of reactive oxygen species (ROS), and increased production of matrix metalloproteinases (MMPs), including MMP-3, in synoviocytes that result in joint destruction. There is limited information on how proteolytic enzymes such as MMP-3 can be regulated. We evaluated the effect of the antioxidant N-acetylcysteine (NAC) on RA and identified the relationship between the c-Jun N terminal kinase (JNK) pathway and MMP-3. We hypothesized that elucidating this relationship would lead to novel therapeutic approaches to RA treatment and management. METHODS: We investigated the effect of administering a low dose (1000 μM or less) of an antioxidant (NAC) to human rheumatoid fibroblast-like synoviocytes (MH7A cells). We also investigated the response of antioxidant genes such as nuclear factor erythroid -derived 2-related factor 2 (Nrf2) and Sequestosome 1 (p62). The influence of MMP-3 expression on the JNK pathway leading to joint destruction and the mechanisms underlying this relationship were investigated through primary dispersion culture cells collected from the synovial membranes of RA patients, consisting of rheumatoid arthritis-fibroblast-like synoviocytes (RA-FLS). RESULTS: Low-dose NAC (1000 μM) increased the expression of Nrf2 and phospho-p62 in MH7A cells, activating antioxidant genes, suppressing the expression of MMP-3, and inhibiting the phosphorylation of JNK. ROS, MMP-3 expression, and IL-6 was suppressed by administering 30 μM of SP600125 (a JNK inhibitor) in MH7A cells. Furthermore, the administration of SP600125 (30 μM) to RA-FLS suppressed MMP-3. CONCLUSIONS: We demonstrated the existence of an MMP-3 suppression mechanism that utilizes the JNK pathway in RA-FLS. We consider that the JNK pathway could be a target for future RA therapies.

DOI： 10.1186/s13018-020-01595-9

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記述言語：英語   掲載種別：研究論文（学術雑誌）  

BACKGROUND: Primary osteosarcoma in elderly patients are rare malignant tumors. Its optimal treatment has not yet been determined. METHODS: This retrospective study included 104 patients aged >50 years with resectable, non-metastatic osteosarcoma treated by the members of the Bone and Soft Tissue Tumor Study Group of the Japan Clinical Oncology Group. The effects of adjuvant chemotherapy were estimated by comparing outcomes in patients who received surgery plus chemotherapy with those who underwent surgery alone. RESULTS: Median age at presentation was 59 years. Neoadjuvant and adjuvant chemotherapy was administered to 83 (79.8%) patients. Patients who underwent surgery plus chemotherapy and those who underwent surgery alone had 5-year overall survival (OS) rates of 68.6% and 71.7%, respectively (p = 0.780), and 5-year relapse free survival (RFS) rates of 48.2% and 43.6%, respectively (p = 0.64). Univariate analysis showed that resection with wide margins was significantly correlated with better prognosis. CONCLUSIONS: The addition of chemotherapy to surgery did not improve OS or RFS in patients aged >50 years with resectable, non-metastatic osteosarcoma. Surgery with wide margins was only significantly prognostic of improved survival. The effect of chemotherapy in elderly osteosarcoma patients was unclear.

DOI： 10.1016/j.jos.2019.04.008

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記述言語：英語   掲載種別：研究論文（学術雑誌）  

Stanniocalcin-1 (STC1) is a glycoprotein that was originally identified as a calcium-regulating hormone in bony fish, and that has been shown to also critically mediate cell growth, proliferation and differentiation, etc. in humans. Increased STC1 expression levels have been previously detected in different human cancer samples, such as those isolated from lung, breast, ovary, colon, pancreas, and liver tumors; thus, the present study evaluated STC1 expression in various soft-tissue tumors. STC1 mRNA isolated from 16 cell lines and 186 clinical soft-tissue tumor specimens were analyzed via quantitative real-time PCR, and the calculated expression levels were normalized to those exhibited by STC1-expressing MDA-MB-231 cells. The results of these analyses did not reveal any specific histological tumor types that displayed significantly increased STC1 expression; however, they did not indicate that STC1 expression was significantly higher in malignant compared to benign soft-tissue tumors. Furthermore, in adipocytic tumors, STC1 expression in dedifferentiated liposarcomas was found to be highest and lowest in lipoma tissues, respectively, suggesting that adipocytic tumors may express increasely high levels of STC1 mRNA as they become histologically more advanced. STC1 expression correlates with the malignancy grade in soft-tissue tumors.

DOI： 10.18999/nagjms.82.1.85

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：NAGOYA UNIV, SCH MED  

Stanniocalcin-1 (STC1) is a glycoprotein that was originally identified as a calcium-regulating hormone in bony fish, and that has been shown to also critically mediate cell growth, proliferation and differentiation, etc. in humans. Increased STC1 expression levels have been previously detected in different human cancer samples, such as those isolated from lung, breast, ovary, colon, pancreas. and liver tumors; thus, the present study evaluated STC1 expression in various soft-tissue tumors. STC1 mRNA isolated from 16 cell lines and 186 clinical soft-tissue tumor specimens were analyzed via quantitative real-time PCR, and the calculated expression levels were normalized to those exhibited by STC1-expressing MDA-MB-231 cells. The results of these analyses did not reveal any specific histological tumor types that displayed significantly increased STC1 expression; however, they did not indicate that STC1 expression was significantly higher in malignant compared to benign soft-tissue tumors. Furthermore, in adipocytic tumors, STC1 expression in dedifferentiated liposarcomas was found to be highest and lowest in lipoma tissues, respectively, suggesting that adipocytic tumors may express increasely high levels of STC1 mRNA as they become histologically more advanced. STC1 expression correlates with the malignancy grade in soft-tissue tumors.

DOI： 10.18999/nagjms.82.1.85

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記述言語：日本語   出版者・発行元：日本レックリングハウゼン病学会  

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記述言語：英語   掲載種別：研究論文（学術雑誌）  

BACKGROUND: Patellar tendon autograft after intraoperative extracorporeal radiotherapy has been used for reconstruction of the extensor mechanism following limb-sparing wide tumor resection around the knee. The purpose of this study was to determine the clinical outcome of this reconstruction technique. METHODS: We retrospectively reviewed six consecutive patients with peripatellar tendon and proximal tibial sarcoma who underwent reconstruction of the knee extensor mechanism. The resection area was planned to be contained with the patellar tendon in order to obtain a wide margin. First, the patella was osteotomized at the midline, and the inferior half of patella, patellar tendon, and tibial tuberosity were excised en bloc. The resected segments were devitalized with intraoperative extracorporeal radiotherapy and reimplanted into the original site. A follow-up evaluation included an assessment of the range of motion, extensor lag, the International Society of Limb Salvage score, and complications. RESULTS: Six patients were followed up for 121-270 months. One patient underwent an additional reconstruction with total knee arthroplasty due to a collapse of the tibial subchondral bone. A supracondylar fracture of the femur occurred in two patients, and a delayed union of the osteosynthesis site of the tibial shaft was observed in one patient. At the latest follow up, extensor lag had a median of five degrees, and International Society of Limb Salvage scores had a median of 83%. No local recurrence or rupture of the patellar tendon was observed. CONCLUSIONS: Reconstruction of the knee extensor mechanism using a patellar tendon treated with intraoperative radiotherapy is a reliable and successful method.

DOI： 10.1016/j.knee.2019.10.008

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DOI： 10.1038/s41598-019-56265-6

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記述言語：日本語   出版者・発行元：(一社)日本小児血液・がん学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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掲載種別：研究論文（学術雑誌）  

DOI： 10.1620/tjem.248.87

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記述言語：英語   掲載種別：研究論文（学術雑誌）  

DOI： 10.1016/j.jtcvs.2019.04.015

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DOI： 10.1089/jpm.2018.0415

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記述言語：英語   掲載種別：研究論文（学術雑誌）  

DOI： 10.1371/journal.pone.0215336

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記述言語：英語  

A 34-year-old woman was diagnosed with acute promyelocytic leukemia. Chemotherapy was administered following the JALSG APL204 protocol. Induction therapy with all-trans retinoic acid resulted in complete remission on day 49. She developed coccygeal pain from day 18, which spread to the spine and cheekbones and lasted 5 weeks. She had similar bone pain on days 7-10 of the first consolidation therapy and on days 4-12 of the second consolidation therapy. Oral loxoprofen was prescribed for pain relief. On day 33 of the third consolidation, white blood cell and neutrophil counts were 320/μL and 20/μL, respectively. After she developed epigastralgia and hematemesis, she developed septic shock. Gastroendoscopy revealed markedly thickened folds and diffusely damaged mucosa with blood oozing. Computed tomography revealed thickened walls of the antrum and the pylorus. Despite emergency treatments, she died. Bacterial culture of the gastric fluid yielded Enterobacter cloacae and enterococci growth. Collectively, she was diagnosed with phlegmonous gastritis. Retrospective examination of serial bone marrow biopsy specimens demonstrated progressive bone marrow fibrosis, which may have caused prolonged myelosuppression. Thus, evaluation of bone marrow fibrosis by bone marrow biopsy after each treatment cycle might serve as a predictor of persistent myelosuppression induced by chemotherapy.

DOI： 10.1155/2019/1616237

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記述言語：英語   掲載種別：研究論文（学術雑誌）  

Erdheim-Chester disease is a rare histiocytosis with insufficient clinical data. To clarify the clinical features and prognostic factors of Erdheim-Chester disease, we conducted a nationwide survey to collect the detailed data of 44 patients with Erdheim-Chester disease in Japan. The median age of onset of the participants was 51 (range: 23-76) years, and the median number of involved organs per patient was 4 (range: 1-11). The existence of central nervous system disease was correlated with older age (P=0.033), the presence of cardiovascular lesions (P=0.015), and an increased number of involved organs (P=0.0042). The median survival from the onset was 10.4 years, and >3.0 mg/dL C-reactive protein level at onset was associated with worse outcome (median survival, 14.6 vs. 7.4 years; P=0.0016). In a multivariate analysis, age >60 years (hazard ratio, 25.9; 95% confidence interval, 2.82-237; P=0.0040) and the presence of digestive organ involvement (hazard ratio, 4.74; 95% confidence interval, 1.05-21.4; P=0.043) were correlated with worse survival. Fourteen patients had available histological samples of Erdheim- Chester disease lesions. BRAFV600E mutation was detected in 11 patients (78%) by Sanger sequencing. A correlation between BRAF mutation status and clinical factors was not observed. Our study revealed that age and digestive organ involvement influence the outcome of Erdheim-Chester disease patients, and an inflammatory marker, such as C-reactive protein, might reflect the activity of this inflammatory myeloid neoplasm.

DOI： 10.3324/haematol.2018.190728

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掲載種別：研究論文（学術雑誌）  

DOI： 10.1016/j.jos.2018.09.009

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記述言語：英語   掲載種別：研究論文（学術雑誌）  

DOI： 10.1111/cas.13769

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記述言語：英語   掲載種別：研究論文（学術雑誌）  

DOI： 10.3892/mco.2017.1489

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：SPRINGER JAPAN KK  

Histiocytic sarcoma, a rare hematopoietic neoplasm with evidence of histiocytic differentiation, is often refractory to conventional chemotherapy and radiotherapy, and its prognosis is generally dismal. The optimal management of this malignancy has not been established. We report a case of 8-year-old girl with histiocytic sarcoma involving the left femur. The tumor rapidly responded to a combination of cladribine and high-dose cytosine arabinoside, an aggressive salvage regimen for refractory Langerhans cell histiocytosis, and became impalpable during the first cycle. The patient has remained in complete remission more than 7 years from diagnosis.

DOI： 10.1007/s12185-017-2202-8

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

2005〜2015年までに、無治療のまま1ヵ月以上の間隔で複数回の画像評価を行った骨・軟部肉腫症例20例(男性14例、女性6例、年齢14〜85歳)を対象に、CTあるいはMRIのDICOMで腫瘍倍加時間を測定した。その結果、組織診断は未分化多型肉腫4例、高分化型脂肪肉腫、悪性末梢神経鞘腫瘍、平滑筋肉腫が各2例、その他10例で、骨腫瘍3例、軟部腫瘍17例であった。倍加時間は23〜858日、推定腫瘍発生時期は2.4〜84.7年前、推定腫瘍発生年齢は-8〜79歳であった。4例は先天性発生と推定され(生下時3例、-8歳1例)、そのうち2例は高齢者であった。最終転帰は腫瘍死9例、有病生存4例、無病生存7例で、腫瘍倍加時間が100日以内の6例は腫瘍死5例、多発転移1例で予後不良であった。

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：ELSEVIER SCIENCE BV  

Background: Reconstruction of the extensor mechanism after resection of the proximal tibia is challenging, and several surgical procedures are available. The purpose of this study was to determine the outcome of the fibular transposition technique for reconstruction of the extensor mechanism of the knee after proximal tibial resection.
Methods: We retrospectively reviewed five consecutive patients who underwent resection of the proximal tibia with prosthetic reconstruction and reconstruction of the extensor using fibular transposition between 1997 and 2011. There were two female and three male patients with a mean age of 50 years (range, 27 to 76 years). A follow-up evaluation included both passive and active range of motion, extensor lag, the MSTS score and complications.
Results: Patients were followed up for 93 months (range, 44 to 160 months). The mean extensor lag and active flexion were four degrees (range, 0 to 10 degrees) and 103 degrees (range, 85 to 110 degrees), respectively. The mean MSTS score was 80% (range, 73 to 90%). All patients were able to ambulate without crutches at the latest follow-up.
Conclusions: The utilization of the fibular transposition technique is a simple, reliable, and successful procedure for extensor reconstruction after proximal tibial resection. (C) 2016 Elsevier B.V. All rights reserved.

DOI： 10.1016/j.knee.2016.11.005

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記述言語：日本語   出版者・発行元：(一社)日本小児血液・がん学会  

ランゲルハンス細胞組織球症(LCH)は、ランゲルハンス細胞様細胞(LCH細胞)のモノクローナルな腫瘍性増殖をきたす疾患である。骨病変の頻度が最も高く、軟部組織単独病変は稀である。今回我々は、胸椎の骨融解病変、胸膜病変、および骨病変に連続しない後頸部の軟部組織病変を伴う多臓器型LCHの5歳女児例を経験した。本例は当初、後頸部皮下膿瘍および化膿性脊椎炎の疑いで抗菌薬が投与され、解熱し病変部位の腫脹が軽減消失したことから、結果的に化学療法の同意が得られず経過観察の方針となった。約9ヵ月間で2回の再燃を観察し、いずれも抗菌薬投与のみで解熱し、局所症状も軽減消失した。最終的に多剤併用化学療法を導入したところ、速やかに寛解が得られ、4年間再発なく経過良好である。本例はLCHでは稀な軟部組織病変を伴うのみならず、抗菌薬による一時的効果が観察された点で、LCHの複雑な病態を考察する上で貴重と思われる。(著者抄録)

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その他リンク： https://search.jamas.or.jp/index.php?module=Default&action=Link&pub_year=2017&ichushi_jid=J06030&link_issn=&doc_id=20170907100013&doc_link_id=%2Fex9syoga%2F2017%2F005402%2F013%2F0157-0160%26dl%3D0&url=http%3A%2F%2Fwww.medicalonline.jp%2Fjamas.php%3FGoodsID%3D%2Fex9syoga%2F2017%2F005402%2F013%2F0157-0160%26dl%3D0&type=MedicalOnline&icon=https%3A%2F%2Fjk04.jamas.or.jp%2Ficon%2F00004_2.gif

記述言語：日本語   出版者・発行元：東北整形災害外科学会  

小児骨肉腫治療を行った51例(男児24例、女児27例、初診時平均年齢12.1歳)を対象に、二次がん(SMN)発生状況とその特徴について検討した。平均観察期間13.2年の結果、SMNの発生率は7.8%(4例)であった。SMN発生例は全て女児であり、女児はリスク因子と考えられた。また、全例で疾患特異的な融合遺伝子や染色体転座を認め、化学療法の影響が示唆された。

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：東北整形災害外科学会  

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：BIOMED CENTRAL LTD  

Background: A solitary fibrous tumour (SFT) is an unusual neoplasm typically found in soft tissues. Although SFTs can arise in the bones, they very rarely arise in the vertebral arch. Here, we describe a case of a SFT that arose in the vertebral arch of the first lumbar (L1) spinal vertebrae and mimicked osteosarcoma.
Case presentation: A 49-year-old woman presented with a 2-month history of lower back pain and a lumbar region mass. Magnetic resonance imaging demonstrated a heterogeneously enhanced mass in the L1 vertebral arch. The patient received neoadjuvant chemotherapy, followed by a surgical procedure comprising an anterior spinal fusion and en bloc resection. Histologically, our initial diagnosis was osteosarcoma. The postoperative course was uneventful, and the patient received adjuvant chemotherapy. However, the tumour metastasised to the lung 5 years after the first surgery, and a second surgery was performed for lung tumour resection. The histology of the metastatic lung tumour appeared similar to that of the malignant SFT, and the specimen from the first surgery was re-examined. Immunohistochemically, the tumour was positive for STAT6. Reverse transcription-polymerase chain reaction revealed a NAB2-STAT6 fusion gene, thus confirming our final diagnosis of malignant SFT. The patient died of disease progression 8 years after the first surgery; however, there was no evidence of local recurrence.
Conclusions: Malignant SFT in the vertebral arch is extremely rare and very difficult to distinguish histologically an osteoid from lace-like collagen. STAT6 immunostaining is useful for distinguishing malignant SFTs from other neoplasms. Although it is difficult to completely resect a SFT arising from the spine, we demonstrated the feasibility of an en bloc resection of spinal tumours arising from posterior elements, without local recurrence.

DOI： 10.1186/s12957-017-1161-0

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：SPANDIDOS PUBL LTD  

The prognosis of alveolar soft part sarcoma is poor, despite the slow growth of the tumor. A number of cases with spontaneous regression of this rare tumor have been reported. Although the mechanisms underlying spontaneous regression remain uncertain, local immune reaction may be a possible contributing factor. Immunohistochemical expression of human leukocyte antigen (HLA) class I, cluster of differentiation (CD) 3, CD4, CD8, CD20, CD45, CD56, CD68, CD138 and CD163 were assessed in a series of 10 alveolar soft part sarcomas, and the expression profiles were associated with patients' clinicopathological parameters. Expression of HLA class I was observed in almost all the tumor cells of all cases. CD8(+) cells were identified in all tumors with varying densities. Moderate infiltration of CD8(+) cells was detected in three patients; one of these patients survived with long-term tumor remission. Infiltration of CD10(+), CD20(+), CD56(+) or CD138(+) cells was not revealed in all tumors. Moderate-diffuse infiltration of CD163(+) cells was observed in all tumors. To the best of our knowledge, the present study represents the first report of intratumoral immune cells in alveolar soft part sarcoma. Frequent expression of HLA class I in tumor cells was observed. CD8(+) cells were identified at various densi-sarcoma. Moderate infiltration of CD8(+) cells in patients with a good prognosis may indicate the antitumor effects of immune cells in alveolar soft part sarcoma.

DOI： 10.3892/ol.2017.5696

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：SPRINGER  

Intravertebral cleft (IVC) is a common finding in osteoporotic compression fracture. However, since the vertebral collapse attributable to cancer metastasis is rarely associated with IVC, the phenomenon is generally considered as a sign of a benign lesion. In this study, we retrospectively reviewed the radiographs, computed tomography scans, and magnetic resonance images of 111 patients with spinal metastasis. Three cases (2.7 %) had IVC in the collapsed thoracic vertebral bodies (T7, T8, and T11) attributable to cancer metastasis. IVC alone is not necessarily an indicator of a benign vertebral collapse.

DOI： 10.1007/s00256-016-2505-5

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記述言語：英語   出版者・発行元：SPRINGER JAPAN KK  

DOI： 10.1007/s10147-016-1051-4

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：ELSEVIER SCIENCE INC  

Myoepithelial carcinoma of the breast is an extremely rare tumor composed entirely of malignant spindle cells with myoepithelial differentiation. The majority of previously reported cases have mainly described the clinicopathological features of the disease, and few have presented cytogenetic data. We herein present the case of a 48-year-old woman who was admitted with a left sided breast lump in the inner upper quadrant that was initially diagnosed as a myoepithelioma with potentially malignant disorder. At 12 months after resection, she complained about a newly developed solid mass in the subareolar region of the ipsilateral breast that was diagnosed as an invasive ductal carcinoma. In addition, 16 months after the initial admission, a re-growing remnant lesion recurred in the inner upper quadrant and was ultimately diagnosed as a myoepithelial carcinoma. Lymph node metastasis of the myoepithelial carcinoma was also observed in her left axillary region 11 months after local recurrence. A cytogenetic analysis showed recurring specific chromosomal alterations both in the locally recurrent and in the lymph-node metastatic lesion: 48, XX, t(5;18)(q13;q23),del(6)(q?),+14. + marl. To our knowledge, this is the first published report of clonal chromosomal rearrangements in myoepithelial carcinoma of the breast that presented as metachronic double cancer with invasive ductal carcinoma in the ipsilateral breast.

DOI： 10.1016/j.cancergen.2016.10.005

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

1988年〜2014年の間に施行された腫瘍用下肢人工関節151例中、20年以上にわたり経過観察が可能であった12例を対象に治療成績について検討した。その結果、12例中、7例(58.3%)で平均1.4回の再置換があった。様々な合併症により再置換の必要な場合があったが、歩行機能をはじめとする患肢機能は良好で、患者の満足度も高かった。就業調査では軽作業への配置転換や定年退職があったものの、失業中の症例はなく、社会復帰は順調であった。

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記述言語：英語   出版者・発行元：(一社)日本小児血液・がん学会  

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：SPRINGER JAPAN KK  

Malignant granular cell tumors (MGCTs) are extremely rare neoplasms with only a limited number of studies published to date. The aim of this study is to elucidate the clinicopathological characteristics and prognostic factors of MGCTs.
This is a multi-institutional retrospective study of MGCTs with a central pathological review. A total of 18 cases were retrieved. Specimens were blindly reviewed by two pathologists based on the diagnostic criteria by Fanburg-Smith et al. Kaplan-Meier survival probabilities were calculated, and risk factors for poor prognosis were evaluated.
Three and fifteen cases were diagnosed as atypical GCTs (AGCTs) and mGCTs according to the Fanburg-Smith et al. classification, respectively. Four (one atypical and three malignant) cases had metastasis at the first presentation, including lymph node metastasis. Three out of ten cases treated with wide resection developed local recurrence. Although prolonged static disease periods of a parts per thousand yen1 year were observed in four cases receiving chemotherapy, all cases with local recurrence or metastasis, including two atypical cases, eventually died of disease. The 5- and 10-year overall survival rates for localized MGCTs were 69.2 and 34.6 %, respectively. The presence of necrosis was revealed as a risk factor associated with adverse clinical outcomes.
MGCTs have high rates of recurrence and metastasis including lymph node metastasis. As histologically atypical cases also have metastatic potential, close attention should be paid to AGCTs. The combination of histological evaluation and tumor size may lead to more accurate diagnosis of this rare neoplasm.

DOI： 10.1007/s10147-016-0949-1

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：PUBLIC LIBRARY SCIENCE  

Background
Bedridden patients are at risk of osteoporosis and fractures, although the long-term bone metabolic processes in these patients are poorly understood. Therefore, we aimed to determine how long-term bed confinement affects bone metabolism.
Methods
This study included 36 patients who had been bedridden from birth due to severe immobility. Bone mineral density and bone metabolism markers were compared to the bedridden period in all study patients. Changes in the bone metabolism markers during a follow-up of 12 years were studied in 17 patients aged <30 years at baseline.
Results
The bone mineral density was reduced (0.58 +/- 0.19g/cm(3)), and the osteocalcin (13.9 +/- 12.4 ng/mL) and urine N-terminal telopeptide (NTX) levels (146.9 +/- 134.0 mM BCE/mM creatinine) were greater than the cutoff value for predicting fracture. Among the bone metabolism markers studied, osteocalcin and NTX were negatively associated with the bedridden period. During the follow-up, osteocalcin and parathyroid hormone were decreased, and the 25(OH) vitamin D was increased. NTX at baseline was negatively associated with bone mineral density after 12 years.
Conclusions
Unique bone metabolic abnormalities were found in patients who had been bedridden for long periods, and these metabolic abnormalities were altered by further bed confinement. Appropriate treatment based on the unique bone metabolic changes may be important in long-term bedridden patients.

DOI： 10.1371/journal.pone.0156991

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記述言語：日本語   出版者・発行元：東北整形災害外科学会  

骨芽細胞様細胞である骨肉腫細胞株由来Saos-2とNOS-1を使用し、これまで癌骨転移などで報告されているインターロイキン-6(IL-6)/IL-6受容体(gp130)/STAT経路の活性化によるκ-Bリガンド核内転写因子受容体活性化因子(RANKL)産生のメカニズムが、Saos-2とNOS-1においても同様に作用しているか検討した。Saos-2、NOS-1の両細胞株においてIL-6受容体(gp130)が発現していることは定量的RT-PCR法によって確認できた。細胞培養液へのIL-6添加によりRANKLの発現量はSaos-2とNOS-1の両細胞株において経時的に増加し、特に72時間後には非添加群に比べSaos-2においては約2倍、NOS-1においては約2.5倍高値を示し、IL-6によるRANKLの発現増強効果が認められた。リン酸化特異的な抗体を用いたウエスタンブロット法により、細胞内におけるIL-6受容体の下流シグナルの変化について検討した。IL-6受容体添加後にSaos-2とNOS-1の両細胞株においてリン酸化STAT3の発現が高くなっていることが確認された。しかし、リン酸化AktとERK1/2の発現に大きな変化はみられなかった。Saos-2、NOS-1において、細胞はIL-6受容体を発現し、培養液にIL-6を添加することによりRANKL産生が促進され、そのメカニズムとしてSTAT3の活性化に伴う細胞内シグナルの関与が示唆された。

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：PUBLIC LIBRARY SCIENCE  

The receptor-activator of nuclear kappaB ligand (RANKL) signaling pathway plays an important role in the regulation of bone growth and mediates the formation and activation of osteoclasts. Osteoclasts are involved in significant bone resorption and destruction. Denosumab is a fully human monoclonal antibody against RANKL that specifically inhibits osteoclast differentiation and bone resorption. It has been approved for use for multiple myeloma and bone metastases, as well as for giant cell tumor of bone. However, there is no previous report quantitatively, comparing RANKL expression in histologically varied bone tumors. Therefore, we analyzed the mRNA level of various bone tumors and investigated the possibility of these tumors as a new therapeutic target for denosumab. We examined RANKL mRNA expression in 135 clinical specimens of primary and metastatic bone tumors using real-time PCR. The relative quantification of mRNA expression levels was performed via normalization with RPMI8226, a human multiple myeloma cell line that is recognized to express RANKL. Of 135 cases, 64 were also evaluated for RANKL expression by using immunohistochemistry. Among all of the tumors investigated, RANKL expression and the RANKL/osteoprotegerin ratio were highest in giant cell tumor of bone. High RANKL mRNA expression was observed in cases of aneurysmal bone cyst, fibrous dysplasia, osteosarcoma, chondrosarcoma, and enchondroma, as compared to cases of multiple myeloma and bone lesions from metastatic carcinoma. RANKL-positive stromal cells were detected in six cases: five cases of GCTB and one case of fibrous dysplasia. The current study findings indicate that some primary bone tumors present new therapeutic targets for denosumab, particularly those tumors expressing RANKL and those involving bone resorption by osteoclasts.

DOI： 10.1371/journal.pone.0154680

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：SPRINGER  

The Akt/mTOR and MAPK pathways play important roles in modulating cellular function in response to extracellular signals, and they are known to be activated in certain kinds of sarcomas. Few investigations have examined these pathways in dedifferentiated liposarcoma (DDLS), in relation to clinicopathological features. Clinicopathological and immunohistochemical analyses were conducted using 99 DDLS specimens. An in vitro study was also conducted to examine the antitumor effects of an mTOR inhibitor and a MEK inhibitor on two DDLS cell lines. The clinicopathological analyses revealed that the AJCC staging was a significant prognostic factor for overall survival and that the tumor size, depth, and location were significant prognostic factors for event-free survival. Phosphorylated Akt (pAkt), pmTOR, pS6RP, p4E-BP1, pMEK, and pERK expressions were positive in 57.4, 52.4, 71.4, 57.1, 84.1, and 50.8 % of the dedifferentiated component of the 63 primary DDLSs. Positive staining for pmTOR was significantly more frequent in the dedifferentiated component than the well-differentiated component. A univariate prognostic analysis revealed that pmTOR expression was associated with poor prognosis in the tumors in the retroperitoneum/ventral body cavity. The mTOR and MEK inhibitors dose-dependently inhibited the cell proliferation of both DDLS cell lines and decreased the expression of downstream pS6RP and pERK, respectively. The combined use of the two inhibitors enhanced antiproliferative activity. In conclusion, the Akt/mTOR and MAPK pathways were activated in DDLS specimens, and the inhibition of these pathways decreased cell proliferation in DDLS cell lines. Our findings suggest that these pathways could be a therapeutic target for patients with DDLS.

DOI： 10.1007/s13277-015-4232-2

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：SPANDIDOS PUBL LTD  

A giant-cell tumor of the bone (GCTB) is a benign but locally aggressive bone tumor. Recently, the receptor activator of nuclear factor kappa B (RANK) ligand inhibitor, denosumab, has demonstrated activity against giant-cell tumors. The current study reports a case of a sacral GCTB with lung metastasis. A 19-year-old male patient presented with right buttock pain and right lower leg pain, and a sacral GCTB was diagnosed based on the histological analysis of a biopsy specimen. The patient was successfully treated with neoadjuvant denosumab therapy, which allowed curettage. In addition, the pulmonary nodule reduced in size following denosumab administration, and surgical resection was performed. Since the operation, the patient has been managed with the continued use of denosumab with no sign of recurrence. Microscopic findings from the surgical specimen following denosumab treatment revealed that the giant cells had disappeared and woven bone had formed. The specimen from the pulmonary nodule exhibited similar findings to the surgical specimen. It was reported that denosumab treatment was able to reduce the number of giant cells and RANK-positive stromal cells, and cause the formation of new bone in the primary lesion. The present study reports the first case to demonstrate the efficiency of denosumab in treating pulmonary metastasis of GCTB.

DOI： 10.3892/ol.2015.3858

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：SPANDIDOS PUBL LTD  

Musculoskeletal sarcomas (MSS) are a heterogeneous group of malignancies with relatively high mortality rates. The prognosis for patients with MSS is poor, with few drugs inducing measurable activity. Alkylating agents, namely ifosfamide and dacarbazine, which act nonspecifically on proliferating cells, are the typical therapy prescribed for advanced MSS. A novel alkylating agent, temozolomide (TMZ), has several advantages over existing alkylating agents. TMZ induces the formation of O-6-methylguanine in DNA, thereby inducing mismatches during DNA replication and the subsequent activation of apoptotic pathways. However, due to conflicting data in the literature, the mechanism of TMZ action has remained elusive. Therefore, the present study aimed to evaluate apoptosis in MSS cells treated with TMZ, and to evaluate the correlation between TMZ action and survival pathways, including the phosphoinositide 3-kinase (PI3K)/Akt and extracellular signal-regulated kinase (ERK)1/2 mitogen activated protein kinase (MAPK) pathways. Cell proliferation was evaluated by performing an XTT (sodium 3'-[1-(phenylaminocarbony1)-3,4-tetrazolium] -bis (4-methoxy-6-nitro) benzene sulfonic acid hydrate) assay. Apoptotic morphological changes, for example chromatin condensation, were evaluated by fluorescence confocal microscopy. The expression of the apoptosis-associated proteins caspase-3, poly adenosine diphosphate ribose polymerase (PARP), Akt and ERK1/2, was determined by western blotting. The results of the present study indicated that, in certain MSS cells, the IC50 value was lower than that in TMZ-sensitive U-87 MG cells. Furthermore, TMZ treatment was associated with apoptotic morphological changes and the expression levels of pro-apoptotic cleaved caspase-3 and PARP were also increased in TMZ-treated MSS cells. In addition, the result indicated that PI3K/Akt and ERK1/2 MAPK were constitutively phosphorylated in MSS cells, and phosphorylation of PI3K/Akt was suppressed in certain cells, and maintained in other cells, by TMZ. These observations emphasized the plasticity of MSS cells, and suggested that this plasticity may contribute to the variance in cell sensitivity to TMZ and TMZ-resistance in MSS.

DOI： 10.3892/ol.2015.3506

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記述言語：日本語   出版者・発行元：(株)医学書院  

1999年から2012年までにβ-リン酸3カルシウム(β-TCP)を使用した整形外科手術714例に対し,合併症を含め後ろ向きに調査した.骨腫瘍(263例)に対して使用した症例では術後早期の骨折が3例にみられたが,遅発性の骨折や変形,術後深部感染は生じず良好な成績が得られていた.β-TCP移植を行った脊椎手術(350例)では3例に術後感染が生じ,5例に再固定手術が施行された.関節疾患,骨折に対して移植を行った101例では深部感染は生じなかった.画像判断の可能な例ではいずれも移植β-TCPと周囲骨とは連続性がみられ,症例によっては継時的にほぼすべてが吸収され新生骨に置き換わっていた.β-TCPは吸収されつつ自家骨形成が生じる優れた骨補填材料であり,整形外科手術に用いた場合,感染率の上昇などの危険性は少ないと考えられた.(著者抄録)

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その他リンク： http://search.jamas.or.jp/link/ui/2015404646

記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：SPANDIDOS PUBL LTD  

The present study reports the first case of malignant transformation to osteosarcoma arising from osteochondroma following childhood total-body irradiation (TBI). The association between TBI and later development of osteochondroma is well-known; however, malignant degeneration arising from radiation-induced osteochondroma is rare. The current study describes the case of a 17-year-old boy with osteosarcoma arising from osteochondroma of the left distal humerus, which developed following TBI. TBI was administered as part of a conditioning regimen received prior to autologous peripheral hematopoietic stem cell transplantation (HSCT) at the age of 6 years, following an initial diagnosis of neuroblastoma at the age of 5 years. The patient subsequently underwent preoperative chemotherapy followed by wide local excision and reconstruction with an extracorporeally irradiated autograft. Postoperative chemotherapy was administered, and the patient demonstrated no clinical or radiographic evidence of recurrence after 40 months of follow-up. To the best of our knowledge, this is only the second reported case of malignant degeneration of osteochondroma following childhood TBI, and the first reported case of transformation to osteosarcoma. The current case highlights the importance of close observation for secondary malignancies in this patient population.

DOI： 10.3892/ol.2015.3257

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記述言語：日本語   出版者・発行元：新潟整形外科研究会  

16歳男性。誘因なく労作時左股関節痛が出現した。今回、徐々に増悪し、安静時痛や歩行困難が生じたため近医を受診、単純X線にて骨腫瘍を疑われ、著者らの施設へ紹介となった。臨床症状および画像所見より類骨骨腫と診断され、保存療法を行うも改善が得られず、約半年経過後、CTガイド下にてnidus摘出術と焼灼術を施行した。その結果、術後22日目に患肢部分荷重を開始し、術後83日目の最終観察時には患肢全荷重で日常生活に支障なく、歩行時痛も再発もみられなかった。

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：PUBLIC LIBRARY SCIENCE  

Expression of the tumor suppressor gene TUSC2 is reduced or absent in most lung cancers and is associated with worse overall survival. In this study, we restored TUSC2 gene expression in several wild type EGFR non-small cell lung cancer (NSCLC) cell lines resistant to the epidermal growth factor receptor (EGFR) tyrosine kinase inhibitor erlotinib and analyzed their sensitivity to erlotinib in vitro and in vivo. A significant inhibition of cell growth and colony formation was observed with TUSC2 transient and stable expression. TUSC2-erlotinib cooperativity in vitro could be reproduced in vivo in subcutaneous tumor growth and lung metastasis formation lung cancer xenograft mouse models. Combination treatment with intravenous TUSC2 nanovesicles and erlotinib synergistically inhibited tumor growth and metastasis, and increased apoptotic activity. High-throughput qRT-PCR array analysis enabling multi-parallel expression profile analysis of eighty six receptor and non-receptor tyrosine kinase genes revealed a significant decrease of FGFR2 expression level, suggesting a potential role of FGFR2 in TUSC2-enhanced sensitivity to erlotinib. Western blots showed inhibition of FGFR2 by TUSC2 transient transfection, and marked increase of PARP, an apoptotic marker, cleavage level after TUSC2-erlotinb combined treatment. Suppression of FGFR2 by AZD4547 or gene knockdown enhanced sensitivity to erlotinib in some but not all tested cell lines. TUSC2 inhibits mTOR activation and the latter cell lines were responsive to the mTOR inhibitor rapamycin combined with erlotinib. These results suggest that TUSC2 restoration in wild type EGFR NSCLC may overcome erlotinib resistance, and identify FGFR2 and mTOR as critical regulators of this activity in varying cellular contexts. The therapeutic activity of TUSC2 could extend the use of erlotinib to lung cancer patients with wildtype EGFR.

DOI： 10.1371/journal.pone.0123967

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記述言語：日本語   出版者・発行元：東北整形災害外科学会  

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：SPRINGER  

Soft tissue tumors arising in deep veins of the extremities are uncommon, although a few cases of synovial sarcoma or leiomyosarcoma arising in the femoral vein have been documented. However, to the best of our knowledge, an extraskeletal myxoid chondrosarcoma (EMC) arising in the femoral vein has not been reported in the English literature. We report a case of EMC arising in the femoral vein of a 70-year-old man who presented with right leg edema and was diagnosed with a deep venous thrombosis (DVT) by computed tomography (CT). Magnetic resonance imaging (MRI) revealed a mass in the right proximal thigh that was diagnosed as myxomatous sarcoma by aspiration cytology, and anticoagulant therapy was initiated. The mass was surgically resected en bloc, including the femoral vein and surrounding soft tissue, and the femoral artery was preserved. The femoral vein was not reconstructed. The histologic diagnosis was an extraskeletal myxoid chondrosarcoma. The patient received postoperative local radiation treatment, with a total dose of 60 Gy, and is currently doing well with no evidence of local recurrence or metastasis at 8 months after surgery. In summary, this case report shows that EMC can arise in the femoral vein, and that reconstruction of the femoral vein is not always necessary during surgery for soft tissue tumors.

DOI： 10.1007/s00256-014-1897-3

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記述言語：英語   出版者・発行元：LIPPINCOTT WILLIAMS & WILKINS  

DOI： 10.1212/WNL.0000000000000714

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：SPANDIDOS PUBL LTD  

Secondary osteosarcoma from fibrous dysplasia (FD) is very rare. The etiology of FD is linked to activating missense mutations of the guanine nucleotide-binding protein alpha-subunit (GNAS) gene, which encodes the stimulatory alpha subunit of the G protein (G(s)alpha) and is located at chromosome 20q13. These mutations are central to the pathogenesis of FD; however, it is not known whether G(s)alpha mutations are retained following malignant transformation in FD. In addition, to the best of our knowledge, no studies have been performed on chromosomal analysis of secondary osteosarcoma from FD. The present study presents a case of secondary osteosarcoma arising from polyostotic FD in a 72-year-old male. Chromosomal analysis showed 44, X, -Y, add(4)(p11), add(5)(p15), der(11)add(11)(p15) t(1;11)(q21;q23), add(12)(q11), -13, der(22)t(12;22)(q11;p12). Reverse transcription-polymerase chain reaction (RT-PCR) analysis demonstrated the presence of a G(s)alpha mutation in both the primary tumor cells and secondary osteosarcoma cells. There was no alteration in this mutation in the region of malignant transformation, which suggests that this mutation may be a useful clinical marker for distinguishing de novo osteosarcoma (primary osteosarcoma) from secondary osteosarcoma arising from FD.

DOI： 10.3892/ol.2014.2082

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記述言語：日本語   出版者・発行元：新潟整形外科研究会  

2006年〜2012年に骨腫瘍の疑いで受診し、最終診断が骨髄炎であった6例(男児3例、女児3例、9〜17歳)を対象に、骨腫瘍と骨髄炎の鑑別について検討した。骨髄炎は高熱や局所の腫脹疼痛、血液検査でのWBC、CRP上昇といった強い炎症反応を伴うものから、これらがほとんどみられないものまで臨床症状は多彩である。炎症反応を伴わないあるいは軽微な場合に骨腫瘍との鑑別にしばしば苦労する。鑑別点は好発部位は骨髄炎は長管骨骨幹端、骨腫瘍は症例により多彩、MRIで骨髄炎は周囲のT2高信号、変化が強い、骨腫瘍は骨内外に腫瘤性病変を形成しやすい等である。いずれも迅速な診断が必須で、開放生検、細菌培養を速やかに施行することが重要である。

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：BIOMED CENTRAL LTD  

Background: Diagnosing adipocytic tumors can be challenging because it is often difficult to morphologically distinguish between benign, intermediate and malignant adipocytic tumors, and other sarcomas that are histologically similar. Recently, a number of tumor-specific chromosome translocations and associated fusion genes have been identified in adipocytic tumors and atypical lipomatous tumors/well-differentiated liposarcomas (ALT/WDL), which have a supernumerary ring and/or giant chromosome marker with amplified sequences of the MDM2 and CDK4 genes. The purpose of this study was to investigate whether quantitative real-time polymerase chain reaction (PCR) could be used to amplify MDM2 and CDK4 from total RNA samples obtained from core-needle biopsy sections for the diagnosis of ALT/WDL.
Methods: A series of lipoma (n = 124) and ALT/WDL (n = 44) cases were analyzed for cytogenetic analysis and lipoma fusion genes, as well as for MDM2 and CDK4 expression by real-time PCR. Moreover, the expression of MDM2 and CDK4 in whole tissue sections was compared with that in core-needle biopsy sections of the same tumor in order to determine whether real-time PCR could be used to distinguish ALT/WDL from lipoma at the preoperative stage.
Results: In whole tissue sections, the medians for MDM2 and CDK4 expression in ALT/WDL were higher than those in the lipomas (P < 0.05). Moreover, karyotype subdivisions with rings and/or giant chromosomes had higher MDM2 and CDK4 expression levels compared to karyotypes with 12q13-15 rearrangements, other abnormal karyotypes, and normal karyotypes (P < 0.05). On the other hand, MDM2 and CDK4 expression levels in core-needle biopsy sections were similar to those in whole-tissue sections (MDM2: P = 0.6, CDK4: P = 0.8, Wilcoxon signed-rank test).
Conclusion: Quantitative real-time PCR of total RNA can be used to evaluate the MDM2 and CDK4 expression levels in core-needle biopsies and may be useful for distinguishing ALT/WDL from adipocytic tumors. Thus, total RNA from core-needle biopsy sections may have potential as a routine diagnostic tool for other tumors where gene overexpression is a feature of the tumor.

DOI： 10.1186/1471-2407-14-468

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：日本レックリングハウゼン病学会  

神経線維腫症1型(NF1)に発症した悪性末梢神経鞘腫瘍MPNSTは予後不良の疾患でしばしば全身性に転移を生じ、抗がん剤抵抗性であるといわれている。我々の施設において治療を行ったNF1に合併したMPNSTの23症例に対して診療録を利用した後ろ向き調査を行った。症例は男13例、女10例、治療開始年齢は15から81歳(平均38歳)、腫瘍径は2-25cm(平均13cm)、症状発生から治療開始までは1-144ヵ月(平均30ヵ月)であった。初回外科治療として広範切除がなされた10例には局所再発がなく、辺縁切除となった(前医を含む)9例のうち8例に局所再発がみられた。抗がん剤治療は13例に対して施行され、効果判定可能であった11例では最高効果時の評価で、部分寛解3例、不変1例、進行7例であった。不変以上の効果が得られたものはいずれも大量イホマイドを主体とするレジメンであったが、いずれも再増悪が生じた。最終転帰は腫瘍死14、持続的無病生存6、有病生存2、他因死1でカプランマイヤー法による累積5年生存率は35%であった。大量イホマイドを主体とする化学療法は至適投与量を維持できれば腫瘍の縮小効果が得られる場合があり、多発肺転移の一時的消失や大幅な縮小がみられる症例も存在する。しかし経過とともに抗がん剤不応性になり、転移巣の長期間コントロールを得ることは極めて困難である。(著者抄録)

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記述言語：日本語   出版者・発行元：(NPO)日本小児血液・がん学会・(NPO)日本小児がん看護学会・(公財)がんの子供を守る会  

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記述言語：英語   掲載種別：研究論文（学術雑誌）  

This study investigated the pathway underlying the antitumor activity of telomelysin, a telomerase-dependent, replication-selective oncolytic adenovirus, in soft tissue sarcoma cells. Treatment with telomelysin alone resulted in simultaneous induction of apoptosis and autophagy, whereas cotreatment with telomelysin and 3-methyladenine significantly reduced cell viability and increased apoptosis and the cellular ATP level compared to treatment with telomelysin alone, indicating that telomelysin-mediated autophagy is a death-protective but not death-promoting process. Cotreatment with Z-Val-Ala-Asp-CH2F significantly increased cellular ATP depletion compared to telomelysin-alone treatment while inhibiting telomelysin-induced apoptosis and having no significant effect on cell viability, indicating that it promotes transition from apoptotic to necrotic cell death. © 2013 Japanese Cancer Association.

DOI： 10.1111/cas.12208

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記述言語：日本語   出版者・発行元：東北整形災害外科学会  

尖足拘縮を生じた腓腹筋内血管腫1例と膝屈曲制限を生じた大腿四頭筋内血管腫1例について報告した。症例1は11歳男で、左腓腹部筋外側頭の圧痛と硬結を認め、膝伸展位での左足関節可動域は-45°であった。MRIでは左腓腹筋外側頭にT1強調画像で筋肉と等信号と高信号が混在し、T2強調画像では高信号と低信号が混在する腫瘍があり、管腔構造の造影効果を認めた。症例2は37歳男で、右大腿前面の硬結と圧痛、右膝関節可動域制限があり、MRIでは中間広筋内にT1強調画像で等信号、T2強調画像で高信号、内部に管腔構造の造影効果を伴う腫瘍を認めた。いずれも周囲筋肉と血管腫の切除により関節可動域は改善し、病理組織所見では周囲筋組織の線維化を認め、筋肉内血管腫と診断された。

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記述言語：日本語   出版者・発行元：新潟整形外科研究会  

37歳男性。左鼠径部から臀部側腹部にかけて18×9×2cm大の可動性不良な軟腫瘤を皮下に認め、軟部肉腫と診断され、広範切除術が施行された。その際、鼠径部は大腿動静脈神経の露出および24×17cmの皮膚軟部組織欠損を生じたため、有茎腹直筋皮弁を挙上移行し、左下腹部から左鼠径部の欠損を被覆した。また、左側腹部の腹膜露出部に対しては左大腿直筋弁、左大腿筋膜、左大腿筋膜張筋を左外側大腿回旋動静脈を茎とし、一塊として挙上後、腹膜側が筋膜側に接するように翻転し、欠損部を被覆補強した。更に大腿直筋は筋膜とともに腹膜露出部に縫合固定後、筋・筋膜弁上に左大腿からの分層植皮を施行した。その結果、病理診断では粘液線維肉腫もしくは粘液型悪性線維性組織球腫と診断された。術後は再発はなかったが、腹直筋採取部と植皮部の肥厚性瘢痕および下腹部のbulgingが認められた。尚、目下は膝の伸展・歩行に関しては日常生活に不自由を感じていないが、階段の昇りにやや困難を感じている。

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(一社)日本皮膚悪性腫瘍学会  

20歳、男性。2008年5月頃より頭頂部に腫瘤が出現し、近医で切開・吸引されたが、短期間で腫瘤が再増大した。同年8月20日に当科を初診した。表面に凹凸のある約15cm×10cmの腫瘤が頭頂部にあり、血痂や潰瘍を伴い、易出血性であった。病理組織学的に、真皮、皮下組織に小型円形で均一なCD99陽性の腫瘍細胞がシート状に増殖し、RT-PCRでEWS-FLI1、EWS-ERGは陰性であった。免疫学的検索、FISHの結果よりEwing肉腫と診断した。原発巣と所属リンパ節に対する放射線照射と化学療法を行ったが、無効であった。腫瘍の重さ、悪臭と外観が患者のQOLを損なっていたため、Mohs軟膏を用いた腫瘍の硬化療法と、腫瘍減量術を施行した。その結果、手術時の出血量を少量に抑え、腫瘍を約半分の大きさに減量することができ、患者のQOLの改善をみた。Mohs軟膏は根治不能な皮膚悪性腫瘍の姑息治療に有用である。(著者抄録)

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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その他リンク： http://search.jamas.or.jp/link/ui/2012161645

記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：SPANDIDOS PUBL LTD  

Polyostotic fibrous dysplasia (PFD) is a high-turnover bone disease that frequently entails chronic bone pain, pathological fractures and severe deformities. Recently, bisphosphonates have shown effective antiresorptive properties in the treatment of children or adults with PFD. We report on a 79-year-old female with PR), who had severe lower limb deformity and chronic bone pain in multiple sites of her extremities for more than 55 years. The patient experienced significant decrease in bone pain and bone turnover markers following long-term (8.5 years) treatment with a low-dose oral alendronate treatment (5 mg/day). To the best of our knowledge, this is the first report of a long-term follow-up of a postmenopausal elderly patient with long-standing symptomatic PFD following continuous low-dose oral alendronate therapy. This case report indicates that long-term daily administration of low-dose alendronate alone is a potential treatment option for elderly patients with PFD, particularly those with long-standing bone pain.

DOI： 10.3892/ol.2011.369

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記述言語：日本語   出版者・発行元：(一社)日本癌治療学会  

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：WILEY-BLACKWELL  

We established a dedifferentiated liposarcoma cell line (NDDLS-1) that produces interleukin-6 (IL-6) and granulocyte-colony stimulating factor (G-CSF). The parental tumor showed high leukemoid reactions. The NDDLS-1 cell line was established from a pleural effusion associated with a lung metastasis. Pleomorphic tumor cells arranged in a haphazard growth pattern were seen in xenograft tumors. Numerous inflammatory cells including neutrophils or eosinophils were present throughout the tumor cells. This finding resembled the dedifferentiated area of the parental tumor. The mice bearing NDDLS-1 showed marked leukocytosis. In addition, the NDDLS-1 cells expressed IL-6 and G-CSF at both the mRNA and protein levels, while the NDDLS-1 cells produced near normal levels of tumor necrosis factor alpha (TNF-alpha). In the cytogenetic analysis, both the parental tumor and the NDDLS-1 cells showed a ring or giant marker chromosomes. The NDDLS-1 cell line demonstrated the amplification and expression of both MDM2 and CDK4 by fluorescence in situ hybridization and immunohistochemical analysis. The NDDLS-1 cell line is consistent with the parental dedifferentiated liposarcoma, and it should therefore be useful for further investigations of human dedifferentiated liposarcomas.

DOI： 10.1111/j.1440-1827.2011.02683.x

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：SPRINGER  

A telomerase-specific oncolytic adenovirus, Telomelysin, can selectively kill cancer cells, and be attenuated in normal cells. We herein describe the oncolytic effect of Telomelysin on human osteosarcoma both in vitro and in vivo.
The anti-tumor effects of Telomelysin were evaluated on human osteosarcoma cell lines in vitro and in a mouse xenograft model of human osteosarcoma in vivo. The replication efficiencies of Telomelysin in human osteosarcoma cell lines and normal cell lines and in osteosarcoma xenografts were determined by the expression levels of E1 mRNA and E1A protein using real-time quantitative PCR, Western blot analysis and immunohistochemistry. The in vitro telomerase-specific replication and the viral infection rate were also confirmed by TelomeScan (Telomelysin-GFP), using fluorescent microscopy and flow cytometry, respectively. The cell viabilities were examined by XTT assay, and the tumor volumes were measured every 2 days. The induction of apoptosis was assessed by Western blot analysis, as well as by TUNEL assay.
TelomeScan and Telomelysin were efficiently replicated in human osteosarcoma cell lines and led to a dose- and time-dependent expression of GFP, E1 mRNA and E1A protein. Telomelysin infection induced marked cytolysis and apoptosis in osteosarcoma cell lines in vitro. Neither cytotoxicity nor apoptosis were induced in normal human cell lines. In the human osteosarcoma cell xenograft model, intratumoral injection of Telomelysin resulted in increased viral replication, significant tumor growth suppression and distinct apoptotic cell death.
This study indicated that virotherapy with Telomelysin may provide a promising strategy for the treatment of human osteosarcoma.

DOI： 10.1007/s00432-010-0969-6

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記述言語：日本語   出版者・発行元：東北整形災害外科学会  

膝関節荷重部に発生した骨巨細胞腫26例(男13例・女13例・平均38歳)の治療成績を検討した。初回手術として腫瘍切除または掻爬後に再建術を行い、再発などに対する追加手術を含め最終手術は人工骨充填術13例、セメント充填術8例、固定術3例、腫瘍型人工関節置換術2例であった。観察期間2〜280ヵ月で、再建術後の新たなX線学的関節症性変化の出現は人工骨群1例、セメント群3例に認めた。International Society of Limb Salvage(ISOLS)患肢機能評価で平均点が他の群より低かったのは、painではセメント群4点、functionでは人工関節群3点、emotional acceptanceでは関節固定群1.3点、supportでは人工関節群4点、walkingでは人工関節群3.5点、gaitでは人工関節群3点であった。ISOLS患肢機能評価の総合平均は89%で、人工骨群97%、セメント群85%、関節固定群71%、人工関節群73%であった。人工骨群では、12例中10例が総合100%と良好であった。

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記述言語：日本語   出版者・発行元：東日本整形災害外科学会  

鎖骨周囲に発生した骨軟部腫瘍に対し鎖骨切除術を行った6例について、肩関節自動可動域が正常になるまでの期間とEnnekingの上肢機能評価法による成績を評価した。6例の内5例で8週間以内に関節自動可動域は正常範囲となり、Ennekingの上肢機能評価法でも100%となった。また、6例とも患者満足度は高く、鎖骨切除術の術後成績は良好であった。(著者抄録)

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：SPANDIDOS PUBL LTD  

Podoplanin is a 38 kDa mucin-type transmembrane glycoprotein that was first identified in rat glomerular epithelial cells (podocytes). It is expressed in normal lymphatic endothelium, but is absent from vascular endothelial cells. D2-40 is a commercially available mouse monoclonal antibody which binds to an epitope on human podoplanin. D2-40 immunoreactivity is therefore highly sensitive and specific for lymphatic endothelium. Recent investigations have shown widespread applications of immunohistochemical staining with D2-40 in evaluating podoplanin expression as an immunohistochemical marker for diagnosis and prognosis in various tumors. To determine whether the podoplanin (D2-40) antibody may be useful for the diagnosis of soft tissue tumors, 125 cases, including 4 kinds of benign tumors, 15 kinds of malignant tumors and 3 kinds of tumor-like lesions were immunostained using the D2-40 antibody. Total RNA was extracted from frozen tumor tissue obtained from 41 corresponding soft tissue tumor patients and 12 kinds of soft tissue tumor cell lines. Quantitative real-time PCR reactions were performed. Immunohistochemical and quantitative real-time RT-PCR analyses demonstrated the expression of the podoplanin protein and mRNA in the majority of benign and malignant soft tissue tumors and tumor-like lesions examined, with the exception of alveolar soft part sarcoma, embryonal and alveolar rhabdomyosarcoma, extraskeletal Ewing's sarcoma/peripheral primitive neuro-ectodermal tumor and lipoma, which were completely negative for podoplanin. Since it is widely and highly expressed in nearly all kinds of soft tissue tumors, especially in spindle cell sarcoma, myxoid type soft tissue tumors and soft tissue tumors of the nervous system, podoplanin is considered to have little value in the differential diagnosis of soft tissue tumors.

DOI： 10.3892/or.2011.1141

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：SPANDIDOS PUBL LTD  

Ewing sarcoma/primitive neuroectodermal tumors (ES/PNETs) may arise in bone or soft tissue; however, these tumors rarely originate in the stomach. To the best of our knowledge, only four cases have previously been reported in the English-language literature. A 41-year-old Japanese woman was admitted with abdominal pain and underwent gastrectomy to remove the primary tumor. Immunohistochemistry, chromosomal karyotype and molecular analysis using reverse transcription-polymerase chain reaction were performed in the tumor specimens obtained. Tumor cells showed positive immunoreactivity for CD99, vimentin, CD117 (c-kit), S100, chromogranin A and synaptophysin. The tumor was a gastric ES/PNET with the EWS-FLII fusion gene translocation t(11;22)(q24;q12). Multiple repeat metastasectomies, as well as multi-agent chemotherapy and radiotherapy were performed for recurrent disease. Despite treatment, the patient succumbed due to progressive disease 110 months after the initial surgery for gastric ES/PNET. A review of the reported cases suggests that patients with gastric ES/PNETs have an unfavorable prognosis following resection due to the high propensity of these tumors to metastasize. Thus, multimodal treatment approaches including surgery, as well as multi-agent chemotherapy and radiotherapy may provide a survival benefit for patients with gastric ES/PNETs.

DOI： 10.3892/ol.2011.246

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：INT INST ANTICANCER RESEARCH  

Background: FUS1 is a tumor suppressor gene located on human chromosome 3p21.3. Frequent loss of FUS1 protein expression is associated with lung cancer development. This study examined FUS1 expression and its possible tumor-suppressive role in bone and soft tissue sarcomas. Materials and Methods: The expressions of FUS1 mRNA and FUS1 protein were assessed in sarcoma cell lines, sarcoma tissues, benign bone and soft-tissue tumor (BST) tissues, and healthy tissues. Exogenous FUS1 gene transfection was performed on sarcoma cell lines. Results: FUS1 mRNA expression was detected in all sarcoma cell lines, all benign BSTs and healthy tissues, and almost all sarcoma tissues. In contrast, FUS1 protein expression was frequently lost in sarcoma cells and sarcoma tissues. The exogenous PUS1 gene delivery induced strong FUS1 protein expression, inhibition of cell viability and apoptosis in sarcoma cells. Conclusion: PUS1 may act as a tumor suppressor in bone and soft-tissue sarcomas.

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：SPRINGER TOKYO  

DOI： 10.1007/s00776-010-0001-7

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：MOSBY-ELSEVIER  

DOI： 10.1016/j.jse.2010.05.017

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

表在性の軟部腫瘍や骨外性病変が触知できる骨腫瘍では触診しながら腫瘍または骨外病変を直接穿刺し、通常の骨腫瘍はX線透視下に穿刺を行った。深部の骨・軟部腫瘍に対してはCTガイド下に穿刺を行った。骨腫瘍および軟部腫瘍に対して行われた針生検983例のうち、最終的に切除材料または開放生検で確認ができた穿刺吸引細胞診(FNAC)359例(骨腫瘍90例、軟部腫瘍269例)、針組織生検(CNB)127例(骨腫瘍40例、軟部腫瘍87例)を対象とした。FNACでは、誤診はすべて偽陰性で3例に認めた。感度、特異度、偽陽性率、偽陰性率はそれぞれび94.3%、100%、0%、5%で、正診率は96.7%であった。CNBでは、臨床所見より判定が正しいものと判断された。誤診例はなかった。感度、特異度、偽陽性率、偽陰性率はそれぞれ100%、100%、0%、0%であり、正診率は100%であった。

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記述言語：日本語   出版者・発行元：新潟整形外科研究会  

症例1は30歳女性で、左膝痛が出現し徐々に増悪していたが原因がはっきりしなかった。症状が改善しないため5年後受診し、MRIで血管腫が疑われ受診した。左大腿遠位前面に疼痛・圧痛を認めたが明らかな腫瘤は触知せず、皮膚所見、左膝関節可動域は正常であった。単純X線で特記すべき所見はなく、MRIで左内側広筋内にT1、T2共に筋肉と比べ高信号と低信号が混在する造影効果のある腫瘤を認めた。血管腫を疑い辺縁切除術を施行した。病理診断は筋肉内血管腫で、術後、症状は消失し、3年経過時点で症状の再燃を認めていない。症例2は28歳男性で、左大腿痛が出現し、受診したが原因が分からず、症状は消失と出現を繰り返した。4年後、症状が強くなり、MRIで血管腫を疑われ受診した。左大腿遠位前面に圧痛を認めたが明らかな腫瘤は触知せず、皮膚所見、左膝関節可動域は正常であった。単純X線で特記すべき所見はなく、MRIで左内側広筋内にT1で筋肉と比べやや高信号、T2で筋肉と比べ高信号を呈する造影効果のある内部不均一な腫瘤を認めた。血管腫を疑い辺縁切除術を施行した。病理診断は静脈型血管腫で、術後、症状は消失した。

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：SPRINGER  

We report here, our experience of seven patients with epithelioid sarcomas and their serum CA 125 levels, as well as the results of an in vitro and in vivo study of CA 125 expression in epithelioid sarcoma cells and xenografts using three epithelioid sarcoma cell lines.
In the clinical study, the serum CA 125 levels of seven epithelioid sarcoma patients were examined at multiple time points. Expression of the MUC16 gene that encodes the CA 125 sequence was examined using RT-PCR methods in three epithelioid sarcoma cell lines, FU-EPS-1, SFT-8606 and NEPS, and the CA 125 protein in each cell lysate was examined by Western blot using anti-CA 125 clone OC125 antibody. The concentration of CA 125 in the conditioned medium of each cell line was also measured.
In five of the seven epithelioid sarcoma patients, CA 125 levels reflected regression and progression of their disease. The CA 125 concentrations in the conditioned medium of FU-EPS-1, SFT-8606 and NEPS cells were 259, 252, and 6 U/ml, respectively. Strong expression of MUC16 mRNA was shown in FU-EPS-1 and SFT-8606 cells: correspondingly, a thick band was observed by Western blot analysis in only FU-EPS-1 and SFT-8606 cells.
We concluded that epithelioid sarcoma cells produce and secrete CA 125 into the blood serum and that the elevation of serum CA 125 correlates with disease progression. Therefore, measuring the serum CA 125 level should provide an useful index for diagnosing and monitoring the course of epithelioid sarcoma.

DOI： 10.1007/s00432-009-0678-1

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：WILEY-BLACKWELL PUBLISHING, INC  

Podoplanin is known as a lymphatic marker because its expression is detected in lymphatic but not vascular endothelium. Podoplanin is also expressed in several normal tissues including osteocytes or osteoblasts. A systematic examination of the podoplanin expression was conducted in normal skeletal tissues and some bone tumor cell lines, and the diagnostic value determined in primary bone tumors. Podoplanin mRNA was expressed at a high level in bone marrow tissue and cartilage, and was upregulated with differentiation to osteoblasts in bone marrow cells. Strong podoplanin expression was seen in osteocytes, chondrocytes, and osteoblasts on immunohistochemistry. Podoplanin mRNA was expressed at a high level in several osteosarcoma and chondrosarcoma cell lines, whereas podoplanin was expressed at a low level in a Ewing's/primitive neuroectodermal tumor cell line. In the clinical samples, osteosarcomas (22/26) expressed podoplanin at various levels. In small cell osteosarcomas (2/2), podoplanin was expressed strongly, although the tissue samples included few diagnostic osteoids. Chondrosarcomas (10/10) expressed podoplanin strongly, and chondroblastomas (5/5) expressed podoplanin moderately, while podoplanin was absent or expressed at low levels in Ewing's sarcomas (0/5), chordomas (0/6) and giant cell tumors of bone (1/7). Therefore, podoplanin may be a sensitive immunohistochemical marker of osteogenic and chondrogenic bone tumors.

DOI： 10.1111/j.1440-1827.2009.02510.x

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：SPANDIDOS PUBL LTD  

The elbow is an uncommon site for malignant bone tumors Surgical options for the reconstruction of the elbow Joint are limited and technically challenging In this study, we describe a patient with osteosarcoma of the proximal ulna treated by wide resection and reconstruction with a combined use of free vascularised fibula graft and extracorporeally irradiated osteochondral graft Ten years after the surgery, the patient is alive, without disease and is able to play golf with no lateral instability or pain of the elbow joint A vascularised fibula, combined with extracorporeally irradiated osteochondral graft with ligamentous repair is one of the options for the treatment of malignant bone tumor of the proximal ulna

DOI： 10.3892/ol_00000024

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記述言語：日本語   出版者・発行元：東日本整形災害外科学会  

ゲムシタビンを中心とした化学療法を行った悪性顆粒細胞腫2例を経験したので報告する。症例1:62歳男性。右鼠径部に腫瘤出現。切除後照射施行するも4年後遠隔転移を認め、ゲムシタビン、パクリタキセルの併用療法を開始した。3ヵ月間はSDであった。症例2:49歳女性。頸髄腫瘍にて発症し切除術施行するも、1年後に再発、転移した。再度切除術を施行し、術後照射後にゲムシタビンを中心とした化学療法を開始した。12ヵ月間SDである。(著者抄録)

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記述言語：日本語   出版者・発行元：整形外科バイオマテリアル研究会  

下肢骨端の良性腫瘍に対する掻爬とβ-リン酸三カルシウム(β-TCP)移植の効果を後ろ向きに調べた。患者10名(男7名、女3名、手術時平均29歳)を対象とした。組織型は骨巨細胞腫が7名、軟骨芽細胞腫が3名であった。まず病巣に大きな開窓を施し、病巣を掻爬後、アルゴンビームレーザーと電気メスで掻爬壁を焼灼した。その後骨髄液を添加したβ-TCPを移植した。大腿骨頭例には血管付腸骨移植を、骨皮質の欠損した膝関節症例には遊離腸骨移植を実施した。手術後に再発を認めず、いずれも術後平均3ヵ月目には移植β-TCPと周囲正常骨の境界が不明瞭となり、移植β-TCPは自家骨へと置換されていた。関節可動域は健側と同程度に回復し、両側変形性膝関節症の既往を有する1名以外は疼痛もなく日常生活を送っている。以上より、下肢関節周辺に発生した骨腫瘍に対するβ-TCP移植は長期に安定した成績を期待できる治療法である。

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記述言語：日本語   出版者・発行元：新潟整形外科研究会  

歩行中に右股関節痛が出現した44歳女性症例について検討した。26歳時にIgM型骨髄腫、41歳時にマクログロブリン血症とそれに伴うアミロイドーシスの既往歴があった。単純X線像では右大腿骨頸部骨折および両側大腿骨頸部骨透亮像を認めた。MRI T1強調像では、両側大腿骨頸部および周囲筋内の低信号病変を認めた。画像所見、既往歴からアミロイド沈着による病的骨折の可能性が疑われ、両側大腿骨に人工骨頭置換術、経皮的ピンニング術を行った。摘出標本はアミロイド沈着陽性であった。M蛋白血症を合併する全身性アミロイドーシス患者の骨折は、アミロイド沈着による病的骨折の可能性を考慮すべきであると思われた。

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：ELSEVIER SCIENCE INC  

Pleomorphic rhabdomyosarcoma (PRMS) is a rare variant of rhabdomyosarcoma that occurs mostly in adults. A few cytogenetic studies of PRMS have been reported, but no consistent specific chromosome aberrations were detected. We herein report a cytogenetic study of three cases of pleomorphic rhabdomyosarcoma using a conventional G-banded karyotyping analysis. The three cases appeared to exhibit an extremely complex karyotype with numeric and structural rearrangements. Although the three cases displayed several common aberrations, including -2, -4, -9, -13, -14, -15, -19, -2 1, add(X)(p11), add(1)(q11), add(7)(p11), and add(13)(p11), no recurrent characteristic chromosomal aberrations could be detected. In addition, among these cases and seven other cases of previously reported PRMS, the most frequent chromosomal alterations were -2, -13, -14, -15, -16, and -19. No obviously consistent structural alterations can be found in these 10 PRMS cases, however, thereby suggesting that it is difficult to confirm whether these complex karyotypes correlated with the diagnosis or clinical outcome in PRMS. In this study, we detected MyoDt and myogenin gene transcripts at the mRNA level in four cases of PRMS together with other soft-tissue sarcomas, including seven cases of malignant fibrous hitiocytoma, five cases of liposacroma, and three cases of leiomyosacroma using a real-time quantitative reverse-transcriptase polymerase chain reaction (RT-PCR) analysis. High-level expressions of MyoD1 and myogenin gene transcripts were determined in all cases of PRMS. In contrast, the other non-PRMS sarcomas showed either no expression or extremely weak expressions for both genes. Our findings suggest that the detections of MyoD1 and myogenin transcripts using real-time quantitative RT-PCR, combined with immunohistochemical stains, are extremely sensitive and useful for the diagnosis of PRMS. (C) 2009 Elsevier Inc. All rights reserved.

DOI： 10.1016/j.cancergencyto.2009.02.011

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：INT INST ANTICANCER RESEARCH  

Background: A giant cell tumor (GCT) of bone is a locally aggressive tumor with a propensity for local recurrence. A characteristic pattern of peripheral bone formation has been described in GCT recurrence in soft tissue, and in some pulmonary metastases from benign GCT. Although the bone formation in GCT in supposedly due to bone morphogenetic proteins (BMPs), the expression pattern of BMPs in GCT has not been well investigated. Materials and Methods: The expression of BMPs in GCT tissues, cultured stromal cells from GCT and osteoclast-like giant cells harvested by laser microdissection (LM), as well as from control osteosarcoma (NOS-1) cells was analyzed using reverse transcriptional-semiquantitative PCR. Results: BMP 2, 3, 4, 5 and 6 were expressed in the GCT tissue. The cultured GCT cells expressed BMP 2, 4, 5 and 6. The osteoclast-like giant cells expressed BMP 2, 3, 5 and 6 and BMP 5 was expressed at the highest level. Conclusion: Both stromal cells and osteoclast-like cells in GCT expressed several kinds of BMPs.

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：BIOMED CENTRAL LTD  

Background: Chondromodulin-1 (ChM1), an endogenous anti-angiogenic factor expressed in cartilage, has been suggested to inhibit invasion of endothelial cells into cartilage. In addition, the ectopic administration of ChM1 has been reported to suppress tumorigenesis in vivo. However, it is unclear whether the anti-tumor effect is due to not only the anti-vascularization effect of ChM1, but also its direct action against oncocytes. In the present study, we sought to determine whether ChM1 has a direct action on tumor cells.
Methods: BrdU incorporation assay was performed on human umbilical vein endothelial cells (HUVECs), normal human dermal fibroblasts (NHDFs), HepG2 cells and HeLa cells in the presence or absence of recombinant human ChM1 (rhChM1). An adenovirus that expresses ChM1, Ad-ChM1, was established and applied to the tumor xenografted in vivo, and to in vitro tumor cells cultured on plates or in soft agar. Cell cycle-related proteins and the phosphorylation of Erk, Akt, and GSK3 beta, the downstream molecules of the extracellular matrix-integrin signaling pathways, in HepG2 cells treated with or without Ad-ChM1 were detected by western blot analysis. Luciferase reporter assays of STAT, GAS, and ISRE, which participate in another cytokine signaling pathway, ware performed in HepG2, HeLa, and HUVEC cells.
Results: ChM1 suppressed BrdU incorporation in HUVECs and in HepG2 cells dose-dependently, but did not suppress BrdU incorporation in NHDFs and HeLa cells cultured on plates. In soft agar, however, ChM1 suppressed the growth of HeLa cells, as well as HepG2 cells. Western blot analyses demonstrated that ChM1 decreased the levels of cyclin D1, cyclin D3, and cdk6 and increased those of p21(cip1) without affecting the phosphorylation levels of Erk, Akt, and GSK3 beta in HepG2 cells. The luciferase reporter assay demonstrated that ChM1 suppressed the transcriptional activities of STAT and GAS but not of ISRE.
Conclusion: ChM1 directly suppressed the proliferation of tumor cells in an anchorage- independent manner. However, ChM1 did not alter the phosphorylation of downstream molecules, at which the signaling pathways through growth factor and cytokine receptors converge with the anchorage-dependent pathway. Our results show that ChM1 has a direct anti-tumor effect; moreover, this effect occurs by inhibiting the STAT signaling pathway.

DOI： 10.1186/1471-2407-9-166

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：ELSEVIER SCIENCE INC  

Alveolar soft part sarcoma (ASPS) is a distinct, rare soft tissue tumor with an unknown histogenesis and a tendency for late widespread metastases to lung, bone, and brain. It is now clear that they are caused by a specific unbalanced translocation, der(17)t(X;17)(p11;q25), which results in the formation of all ASPSCR1-TFE3 (alias ASPL-TFE3) fusion gene. The rearrangement results; ill the expression of chimeric transcripts, which can be identified by means of reverse transcriptase-polymerase chain reaction (RT-PCR). We investigated the histogenesis of ASPS and attempted to detect Circulating ASPS tumor cells in peripheral blood. The immunohistochemical and genetic details of four cases and one cell title of ASPS were examined. An immunohistochemical analysis and RT-PCR did not detect myogenic differentiation gene MYOD1. The sensitivity of nested RT-PCR for detection of circulating ASPS cells was assessed by demonstrating that the tumor cell-associated gene translocation could be detected in 50 tumor cells/2 mL of blood. Clinically, it was detectable in a peripheral blood sample (2 mL) of ASPS patient with distant metastases. The findings suggest that ASPS is not of skeletal muscle origin. ASPS tumor cells in the peripheral blood could be monitored by RT-PCR. (c) 2009 Elsevier Inc. All rights reserved.

DOI： 10.1016/j.cancergencyto.2008.11.014

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：BIOMED CENTRAL LTD  

Background: Multinucleated cells are frequently seen in association with a malignant neoplasm. Some of these multinucleated cells are considered to be neoplastic. The mechanism of neoplastic multinucleation remains unknown, but is considered to be induced by either cell-cell fusion or acytokinetic cell division. Myxofibrosarcoma consists of spindled and pleomorphic tumor cells and bizarre multinucleated giant cells. Some of these multinucleated cells are considered to be neoplastic.
Methods: We studied the mitotic activity of the multinucleated cells by Ki-67 immunohistochemistry, and the dynamics and differentiation by live-cell video microscopy in the two myxofibrosarcoma cell lines to determine whether the mechanism of multinucleation is cell-cell fusion or acytokinetic cell division
Results: A Ki-67 immunohistochemical analysis revealed a high positive rate of multinucleated cells, as well as mononuclear cells, and mitotic ability was shown in the multinucleated cells. In live-cell video microscopy, most of the multinucleated cells were induced via the process of acytokinetic cell division.
Conclusion: The current study indicates that a vulnerability of the cytoskeleton components, such as the contractile ring, causes multinucleation to occur from the telophase to the cytokinesis of the cell cycle.

DOI： 10.1186/1756-9966-28-44

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DOI： 10.1186/1756-9966-28-44

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

切除不能な再発あるいは転移を有する骨・軟部腫瘍症例で、同意が得られた8症例に対しシクロスファミド(CPM)100mgの内服を中心とした外来化学療法を施行しており、その短期成績を報告した。症例により、ドキソルビジン(DOX)の静注を月一回施行した。DOX投与後2、3日は食欲不振を来たすことがあり、吐き気や不快感がある場合は、QOLの維持に主眼を置くためDOX静注は早めに中止した。高齢者の1例、長期の入院抗癌剤治療後の2例は好中球減少が生じてCPMを減量したが、血液毒性は軽度であり、連日投与が可能であった。薬剤投与に関しては外来で十分管理可能であった。投与2ヵ月後では部分奏功1例、安定5例、進行2例であった。部分奏功例では施行後1年間は腫瘍の十分な抑制効果がみられたが、DOXが極量となり、CPM内服単独となって再増悪した。腫瘍進行までの期間は1〜11ヵ月で、最終追跡時3例が有病生存で内服を継続中であり、5例が腫瘍死した。

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記述言語：日本語   出版者・発行元：新潟整形外科研究会  

症例1:49歳男。18歳時に第5頸髄を損傷し、完全四肢麻痺、不全呼吸麻痺となり、気管切開され人工呼吸器管理となった。受傷直後より仙骨部褥瘡を認めており、3年前に褥瘡部に隆起性腫瘤が出現し、徐々に増大した。入院時、仙骨部褥瘡に径約20cmの隆起した腫瘤があり、大量の排膿があった。CTにて右臀部から腸骨、仙骨まで浸潤する腫瘍、両肺に転移巣を認めた。右鼠径リンパ節の吸引細胞診にてリンパ節転移と診断され、腫瘍切除および右股関節離断、右大腿部筋皮弁を用いた欠損部再建を行った。病理診断は扁平上皮癌であった。術後肺転移が増大、呼吸状態悪化し、術後35日に死亡した。症例2:55歳男。21歳時に第4胸髄を損傷し、完全対麻痺となった。受傷直後より仙骨部褥瘡があり、約2年前に褥瘡内左半分に腫瘤性病変を認め、fibrous polyposisの診断で腫瘤切除された。その後、再び褥瘡内に腫瘤が出現し、急激に増大し、CTにて右臀部から骨盤内にかけて巨大な腫瘍性病変を認めた。入院時、仙骨部の褥瘡内部に易出血性の腫瘤があり、大量出血と排膿があった。生検で卵円形、紡錘形の細胞を認め、悪性線維性組織球腫と診断され、腫瘍切除および右半骨盤切除を施行し、大腿部筋皮弁を用いて再建した。術後1ヵ月に肺転移、局所再発を認め、化学療法を行ったが腫瘍は皮膚へ浸潤し、全身状態が悪化して術後56日に死亡した。

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記述言語：日本語   出版者・発行元：(株)医学書院  

胞巣状構造を呈するが特異的遺伝子転座を認めない横紋筋肉腫1例を経験したので報告する.症例は1歳2ヵ月の女児で,針細胞診で横紋筋肉腫と診断し,広範切除を行った.術後組織病理学所見では,胎児型の部分と胞巣状構造が混じったパターンであった.典型的胞巣型と異なり,線維性間質に乏しく,大量の横紋筋芽細胞が混在するのが特徴であった.胞巣型に特異的なt(2;13)またはt(1;13)の転座を認めなかった.術後6年9ヵ月の現在,多発性転移を認め,再発と軽快を繰り返し,現在化学療法中である.(著者抄録)

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その他リンク： http://search.jamas.or.jp/link/ui/2008306918

記述言語：日本語   出版者・発行元：東北整形災害外科学会  

1988〜2006年にかけ著者らにより仙骨切断術が施行され、温存できた神経が術中において肉眼で確認できた仙骨脊索腫6症例を対象に、仙髄神経根と膀胱直腸機能の関係を検討した。その結果、1)S3神経根が膀胱直腸機能の維持に重要であることが確認された。2)両側S2神経根を温存した4例すべてに膀胱直腸障害が認められた。3)尿路変更、人工肛門例を除いた5例の自覚症状は、排尿している時の感覚の減弱、尿意の減弱、夜間の失禁、便秘が多かった。4)膀胱と直腸の知覚が低下し、膀胱収縮が減弱した状態であったことから、主に骨盤神経の障害によるものと考えられた。5)これらのことは骨盤神経障害が陰部神経障害より優位に出現していると考えられる傾向であり、過去に指摘されていない事実であった。6)脊索腫に限らず、腫瘍高位によってS3神経根の温存不可能な場合、術後の膀胱直腸機能障害は必発と考えられ、温存不可能な場合は術前に十分な説明が必要であると思われた。

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記述言語：日本語   出版者・発行元：東北整形災害外科学会  

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：SPRINGER  

We report a case of cystic teratoma of the diaphragm that was considered to be a soft tissue lipoma by the preoperative examination. A magnetic resonance imaging revealed an 8x8 cm well-demarcated, homogeneous mass. On T1- and T2-weighted imaging, the mass was isointense to fat. On the fat-suppressed imaging, the signal from the mass was reduced to the same degree as subcutaneous fat. We performed marginal excision. The tumor contained a large number of hairs and oily liquid; and it was connected to the diaphragm.

DOI： 10.1007/s00256-007-0318-2

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：SPRINGER  

Dedifferentiated chondrosarcoma is a rare, highly malignant variant of chondrosarcoma in which a high-grade sarcoma coexists with a low-grade chondroid tumor. We herein review a case of dedifferentiated chondrosarcoma with an osteosarcoma omit component that occurred in the distal femur of a 38-year-old man. We established the cell line (NDCS-1) from a pleural effusion of the metastatic lung tumor. The cell line was characterized by a the G-banded karyotype, polymerase chain reaction (PCR) single-strand conformation polymorphism analysis, spectral karyotyping, and reverse transcriptase PCR (RT-PCR). The tumor exhibited complex karyotypes and a high frequency of chromosomal amplication with p53 mutation. This tumor revealed an osteoblastic and chondroblastic character in vitro and in severe combined immunodeficiency mice. The expression and phosphorylation of platelet-derived growth factor receptor-beta, which seemed to play a major role in the malignant phenotype of chondrosarcoma, was confirmed by RT-PCR and Western blotting. To our knowledge, this is the first report of the establishment of a human dedifferentiated chondrosarcoma.

DOI： 10.1007/s00428-007-0426-3

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：ELSEVIER SCIENCE INC  

We report on a case of a solitary fibrous tumor that developed in the thigh of an 82-year-old woman. The tumor was composed of areas of high-grade sarcoma and typical solitary fibrous tumor. Its karyotype was: 70,XXX, +X [4],+1 [2],add(1) (p3 6) [4], add( 1) [2],+2 [4],-3 [4],+ 6 [4], add(6) (p11) x 2[4],+7[4],+9[3],-11[4],-12[4],-13[4],add(13)(p11)x2[4],-14[4],+15[4],-16[3],-17[4],-19[4],+ 20,[4],+21[4],+22 [2], +mar 1x2[4][cp4]. (c) 2007 Elsevier Inc. All rights reserved.

DOI： 10.1016/j.cancergencyto.2007.04.016

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記述言語：英語   掲載種別：研究論文（学術雑誌）  

Despite its benign nature, a ganglion can be problematic. We successfully treated a patient with large painful ganglion in his ankle by OK-432 (lyophilized incubation mixture of group A Streptococcus pyogenes of human origin) injection. OK-432 injection seems to be a safe, convenient, and effective alternative to surgical treatment for either symptomatic or recurrent ganglia. © Japan College of Rheumatology 2007.

DOI： 10.1007/s10165-007-0597-4

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：ELSEVIER SCIENCE INC  

Ossifying fibromyxoid tumor (OFMT) is a rare but morphologically distinctive soft-tissue tumor. The histologic origin of this tumor is not clearly known, but its various features suggest a schwannian, neuronal, or chondroid origin. We herein report a case of a typical OFMT that occurred in the shoulder of a 65-year-old man. The karyotype exhibited the following complex numeric and structural aberrations: 42 similar to 46, XY, -Y, add(1)(q42),add(6) (p21),t(10;18)(q26;q11),der(11)t(11; 15) (q23;q15),add(12)(q13),ins(14;?)(q13;?),-15, +mar. Combined with several previously reported studies, these aberrations could not identify a common cytogenetic, abnormality in OFMT. (C) 2007 Elsevier Inc. All rights reserved.

DOI： 10.1016/j.cancergencyto.2007.04.009

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記述言語：日本語   出版者・発行元：新潟整形外科研究会  

40歳男。3歳時よりネフローゼ症候群のため内科で加療中であった。38歳時の胸部X線で右第5肋骨と左第6肋骨に骨硬化性の結節影を指摘され、骨島の疑いで経過観察されたが、40歳時に同結節影に増大傾向があり、当科紹介受診した。胸部X線像の経過をみると、17歳時には結節影を認めなかったが、38歳時では右第5肋骨と左第6肋骨に骨硬化性の結節影を認め、40歳時にはそれらが増大していた。骨盤部X線検査では両腸骨・右仙腸関節部、両恥骨、両坐骨、両大腿骨頸部、右大腿骨転子部に小斑状〜結節状の多発性骨硬化像を認め、胸部CT検査では肋骨・胸椎椎体の骨髄内に散在する結節性骨硬化像を認めた。血液検査ではネフローゼ症候群によると思われる低蛋白血症と軽度低リン血症を認めた。左腸骨の開放生検を行い、病理組織学的に海綿骨内に緻密骨を認め、骨島の所見であった。

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：AMER ASSOC CANCER RESEARCH  

NPRL2 is one of the novel candidate tumor suppressor genes identified in the human chromosome 3p21.3 region. The NPRL2 has shown potent tumor suppression activity in vitro and in vivo and has been suggested to he involved in DNA mismatch repair, cell cycle checkpoint signaling, and regulation of the apoptotic pathway. In this study, we analyzed the endogenous expression of the NPRL2 protein and the cellular response to cisplatin in 40 non-small-cell lung cancer cell lines and found that expression of NPRL2 was significantly and reciprocally correlated to cisplatin sensitivity, with a Spearman correlation coefficient of -0.677 (P < 0.00001). Exogenously introduced expression of NPRL2 by N-[1-(2,3-dioleoyloxyl)propyl]-NNN-trimethylammoniummethyl sulfate: cholesterol nanoparticle-mediated gene transfer significantly resensitized the response to cisplatin, yielding a 40% greater inhibition of tumor cell viability and resulting in a 2- to 3-fold increase in induction of apoptosis by activation of multiple caspases in NPRL2-transfected cells compared with untransfected cells at an equal dose of cisplatin. Furthermore, a systemic treatment with a combination of NPRL2 nano-particles and cisplatin in a human H322 lung cancer orthotopic mouse model significantly enhanced the therapeutic efficacy of cisplatin and overcame cisplatin-induced resistance (P < 0.005). These findings implicate the potential of NPRL2 as a biomarker for predicting cisplatin response in lung cancer patients and as a molecular therapeutic agent for enhancing response and resensitizing nonresponders to cisplatin treatment.

DOI： 10.1158/0008-5472.CAN-06-1483

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：INT INST ANTICANCER RESEARCH  

Background: Activating Gs a mutations have been identified in most instances of fibrous dysplasia (FD). This mutation leads to consistently elevated intracellular cyclic adenosine monophosphate (cAMP) levels, with various biological consequences. The development of secondary sarcoma in FD is a rare but well-established phenomenon. This finding raised the possibility that a common gene mutation exists in these tumors. Materials and Methods: The expression of the Gs a mutation was examined in 16 cell lines and 173 musculoskeletal tumor tissues, including 13 cases of FD, via RT-PCR and sequence analysis. Results: No expression of a Gs a mutation was detected in any cell line or clinical tissue sample, excluding FD tissues. Direct sequence analysis demonstrated results identical to those of RT-PCR. Conclusion: Activating Gs a mutation rarely occurs in musculoskeletal tumors other than FD. The occurrence of most sarcomas displays no correlation with Gs a mutations.

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：ELSEVIER SCI LTD  

Prominent osteoconductive activity and the biodegradable nature of commercially available beta-tricalcium phosphate (beta-TCP, OSferion (R)) have been documented in animal experiments. We analyzed four cases of involving grafted OSferion (R) in human bone with respect to histological features by routine hematoxylin and eosin staining, silver impregnation, immunohistochemistry and in situ hybridization. OSferion (R) 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 (R) 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 (R). (c) 2005 Elsevier Ltd. All rights reserved.

DOI： 10.1016/j.biomaterials.2005.08.034

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：APSIT ASSOC PROM STUD IMMUNOL TUMOR  

The present investigation examined the effect of bisphosphonates on six mesenchymal tumor cell lines and the mechanisms of inhibition of tumor cell proliferation. HT-1080, a fibrosarcoma cell line that exhibits increased Ras activity due to a mutation of the Ras gene, demonstrated significantly reduced tumor cell proliferation upon treatment with incadronate. The other cell lines, however, which lack mutation of the Ras gene, showed no influence upon treatment with incadronate. Autoradiography demonstrated no difference in the uptake of 3 H-labelled incadronate between susceptible and unaffected cells. The anti-proliferation of HT-1080 was reversed by the addition of geranylgeranyl pyrophosphate. Etidronate exhibited no influence on all tested cell lines. On the basis of these data, we hypothesized that incadronate inhibits the mevalonate pathway and blocks oncogenic Ras signaling. In an effort to confirm this hypothesis, the influence of incadronate on an oncogenic Ras transfected BALB/3T3 cell line (Bhas 42) and a parental BALB/3T3 cell line were compared. The parental BALB/3T3 cells showed slight inhibition upon treatment with incadronate, however, the proliferation of Bhas 42 cells was significantly reduced. These results suggest that incadronate suppresses oncogenic Ras-activated mesenchymal tumors through the inhibition of Ras signaling pathways.

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：CHURCHILL LIVINGSTONE INC MEDICAL PUBLISHERS  

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 cernentless 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.

DOI： 10.1016/j.arth.2005.07.004

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：B M J PUBLISHING GROUP  

This report describes a case of congenital dermatofibrosarcoma protuberans (DFSP) with fibrosarcomatous (FS) and myxoid areas. Immunohistochemical results showed that tumour cells in ordinary DFSP areas were diffusely positive for CD34, whereas in the FS and myxoid areas, few tumour cells were positive for this antigen. Ki-67 positive tumour cell numbers were greater in the FS (11.8%) and myxoid areas (19.8%) relative to ordinary DFSP areas (2.2%). Reverse transcription polymerase chain reaction and sequence analysis showed the presence of an identical COL1A1-PDGFB fusion transcript in ordinary DFSP ( plaque-like area), FS, and myxoid areas of DFSP. These results indicate that the three components of DFSP have a common histogenesis. This study documents the first application of gene analysis involving the myxoid area of DFSP.

DOI： 10.1136/jcp.2004.024430

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：ELSEVIER SCIENCE INC  

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 similar to q23 and 8q13-qter regions and loss of the 9p21-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. (c) 2005 Elsevier Inc. All rights reserved.

DOI： 10.1016/j.cancergencyto.2005.02.003

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記述言語：日本語   出版者・発行元：東北整形災害外科学会  

症例1:16歳男.右大腿部腫脹があり,血腫除去術を施行された.術後創部からの出血が持続し,病理検査にて血腫は肉腫と多量の壊死組織であることが判明し,この時点で骨盤内腫瘤は転移巣であり,かつ多発肺転移の存在が判明した.アドリアシン,イフォマイドを施行したが,呼吸不全のため死亡した.最終的に骨外性Ewing肉腫と診断した.症例2:26歳女.後腹膜巨大嚢腫の診断で嚢腫除去術を施行し,術後Ewing肉腫と判明したため多剤併用化学療法を開始した.8年経過し無病生存中である.症例3:34歳男.左胸部皮下腫瘤があり,針生検でEwing肉腫が疑われたため多剤併用化学療法を開始した.1サイクル終了後に広範切除術を行い,現在無病生存中である.症例4:78歳女.左大腿軟部腫瘤があり,大腿動静脈温存腫瘍内切除術を行った.術後Ewing肉腫と判明したが,肺転移による呼吸不全のため死亡した

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：AMER SOC CLINICAL ONCOLOGY  

DOI： 10.1200/JCO.2005.05.042

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記述言語：日本語   掲載種別：研究論文（学術雑誌）  

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：ELSEVIER SCIENCE INC  

Germline mutation and functional loss of EXT1 or EXT2 are commonly found in multiple osteochondrornas and predispose to the development of chondrosarcoma. Mutations of EXT1 and EXT2 have rarely been detected in sporadic secondary chondrosarcomas from osteochondrorna; 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 I 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, I secondary to enchondromatosis, I 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. (c) 2005 Elsevier Inc. All rights reserved.

DOI： 10.1016/j.cancergentyto.2004.08.031

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：BRITISH EDITORIAL SOC BONE JOINT SURGERY  

We describe two patients with a diffuse haemangioma of the lower limb complicated by pathological fracture of the femoral shaft, one of whom was treated by a bone graft and immobilisation in a cast, and the other by external fixation and injection of bone marrow. A review of the literature identified difficulty in control of bleeding and obtaining bony union.

DOI： 10.1302/0301-620X.87B3

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記述言語：英語   掲載種別：研究論文（学術雑誌）  

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 (β-TCP), respectively. Mean follow-up was 97 and 26 months in cases involving HA implantation and β-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 β-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 β-TCP. Highly purified β-TCP appears to be advantageous relative to HA for surgical intervention in bone tumors consequent to the nature of remodeling and superior osteoconductivity. © 2004 Wiley Periodicals, Inc.

DOI： 10.1002/jbm.b.30136

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：WILEY-LISS  

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. (C) 2004 Wiley Periodicals, Inc.

DOI： 10.1002/jbm.b.30136

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：LIPPINCOTT WILLIAMS & WILKINS  

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.

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記述言語：日本語   出版者・発行元：新潟整形外科研究会  

78歳女.左大腿骨骨幹部骨折を受傷し,観血的骨接合術が施行された.しかし皮膚と筋膜の切開のみで3000mlの出血があり,臀部から下肢全体に広がる血管腫と判明した.X線所見で大腿骨骨幹部に横骨折を認め,骨折部周囲の皮下組織内に小さな石灰化を数個認めた.側面像では大腿骨の横径が小さく,骨硬化像を伴っていた.MRI-T2強調画像にて骨を含む大腿部全体に,高信号領域と低信号領域が混在し,血管腫と考えられる病変を認めた.創外固定術を施行し,術中出血は20mlであったが,術後も筋層内への出血と考えられる大腿軟部の腫脹を認めた.濃厚赤血球と新鮮凍結血漿を4単位ずつ輸血したが,術後1週で貧血が残存し,濃厚赤血球と新鮮凍結血漿を2単位ずつ更に輸血した.骨折受傷から約2年後に抜釘術を行い,再骨折防止目的で大ギプスを装着した.抜釘後8ヵ月現在も骨癒合良好で,膝の可動域は伸展0°,屈曲50°で疼痛もなく日常生活を送っている

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：ELSEVIER SCIENCE INC  

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. (C) 2004 Elsevier Inc. All rights reserved.

DOI： 10.1016/j.cancergenyto.2003.11.007

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：OXFORD UNIV PRESS  

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.

DOI： 10.1093/jjco/hyh072

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記述言語：日本語   出版者・発行元：東北整形災害外科学会  

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：AMER ASSOC CANCER RESEARCH  

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.

DOI： 10.1158/1078-0432.CCR-03-0345

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：AMER SOC MICROBIOLOGY  

Ligaments and tendons are comprised of tough yet flexible connective tissue. Little is known, however, about the precise characteristics of the cells in ligaments and tendons due to the absence of specific markers and cell lines. We recently reported a periodontal ligament cell line, PDL-L2, with suppressed Runx2/Osf2 transcriptional activity and an inability to form mineralized nodules. The present study demonstrates that the homeobox protein Msx2 is a key factor in suppressing those two functions. Msx2 colocalizes with Runx2/Osf2 and suppresses its activity cooperatively, acting with another corepressor, TLE1, as a complex to recruit histone deacetylase 1 activity. Reverse transcription-PCR and in situ hybridization demonstrated that Msx2 expression is higher in periodontal ligament and tendon cells than in osteoblasts. Stable reduction of Msx2 expression in PDL-L2 cells induces osteoblastic differentiation, thereby causing matrix mineralization. Conversely, stable, forced Msx2 expression in MC3T3-E1 cells prevented osteoblast differentiation and matrix mineralization. Msx2-induced suppression of osteoblast differentiation was repressed by bone morphogenetic protein 2. In addition, Msx2 was downregulated in a symptom- and calcification-dependent manner at the affected region in patients with ossification of the posterior longitudinal ligament. Our findings indicate that Msx2 plays a central role in preventing ligaments and tendons from mineralizing.

DOI： 10.1128/MCB.24.8.3460-3472.2004

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：SPRINGER  

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.

DOI： 10.1007/s00256-003-0701-6

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：W B SAUNDERS CO  

Liposclerosing myxofibrous tumor (LSMFT) is a benign fibroosseous lesion that is characterized by mixture of histologic elements including lipoma, fibroxanthoma, myxoma, ischemic ossification, and fibrous dysplasia (FD)-like features. These tissue components are seen in the original reports of FD; however, the relationship between LSMFT and FD is not clear. Point mutation of the a subunit of G protein (Gs alpha), which increases cyclic adenosine monophosphate formation, has been recognized as the cause of McCune-Albright syndrome as well as polyostotic and monostotic FD of bone. Gs alpha mutation at the Arg(201) codon in 2 patients of LSMFT was demonstrated in the present study. Although direct sequencing analysis using the fresh-frozen materials could not detect the mutation, the polymerase chain reaction fragmentation length polymorphism (PCR-RFLP) disclosed the missense point mutation Gs a at the Arg201 codon in 2 cases involving LSMFT. This result strongly suggests that a subset of LSMFT is a variant form of FD. (C) 2003 Elsevier Inc. All rights reserved.

DOI： 10.1016/S0046-8177(03)00430-1

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記述言語：日本語   出版者・発行元：新潟整形外科研究会  

症例1:51歳男.主訴は右腋窩部腫瘤.MRIでは尺骨神経と思われる部位にT1強調で筋肉と比し等〜高輝度,T2強調で辺縁が著明に高輝度で,内部がやや低輝度ないわゆるTarget signを呈する多房性の腫瘤が認められた.多発性神経鞘腫を疑い腫瘍核出術を施行した.大小6個の黄白色を呈する腫瘤が摘出され,病理組織診断は良性神経鞘腫であった.腫瘍は紡錘形細胞が密に存在し,充実性のAntoni Aと細胞密度が疎で粘液状のAntoni Bが混在してみられた.症例2:37歳女.主訴は左上肢腫瘤,疼痛.左前腕部のMRIではT1強調で筋肉に比し低輝度,T2強調でTarget sign陽性,T1〜Gdにてよく造影される多房性の腫瘤がみられた.多発性神経鞘腫を疑い腫瘍核出術を施行した.大小6個の黄白色を呈する腫瘤が摘出され,病理組織診断は神経鞘腫であった.本例もAntoni AとAntoni Bが混在してみられた.術後,橈骨神経領域にMMT1〜3程度の筋力低下が出現した.術後1年半経過した現在,筋力はほぼ回復しADL上不自由なくなっている

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：LIPPINCOTT WILLIAMS & WILKINS  

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.

DOI： 10.1097/01.BRS.0000092344.14376.D9

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：NATURE PUBLISHING GROUP  

Adenovirus vectors are expected to be a powerful tool for gene therapy to treat severe fractures. Adenovirus invades cells through binding to the coxsackievirus and adenovirus receptor (CAR) on the cell membrane. CAR expression is low in normal adult animals, but it is induced on regenerating cells in some experimental models. We made a rib fracture model in mice and evaluated the histological changes and CAR mRNA expression by RT-PCR 1, 5,10,14, and 21 days after the fracture. CAR mRNA was expressed exclusively in the fractured ribs at each time point, but not in the normal ribs. We detected the CAR protein immunohistochemically in fibroblast-like cells in the fracture callus on days 10 and 14 after fracture. In situ hybridization showed that these fibroblast-like cells expressed mRNA of type I collagen and osteppontin, but not osteocalcin, defining the cells as immature osteoblasts. We then transferred small doses (10(4)-10(8) PFU) of lacZ-expressing adenovirus vector into immature osteoblasts on day 14. beta-galactosidase was detected only on the immature osteoblasts at every dose. Immature osteoblasts play an important role in the matrix replacement step in fracture healing. CAR-mediated gene transfer into immature osteoblasts can be reasonable for adenovirus-mediated treatment of fracture healing.

DOI： 10.1038/sj.gt.3302060

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：INT INST ANTICANCER RESEARCH  

Local recurrence free survival rate for myxoid liposarcoma (MLS) is related to negative surgical margins. The goal of surgery in the treatment of MLS is to obtain tumor free surgical margins. When a tumor is adjacent to critical structures, wider resection, which can result in amputation, would be selected for local control. While recognizing that marginal or intralesional resection is associated with high risk of recurrence, circumstances occur that lead surgeons to conduct conservative surgery. To determine whether marginal or intralesional resection combined with radiotherapy can achieve local control, 10 cases (12 tumors) were reviewed involving MLS treated with marginal (8 tumors) or intralesional resection (4 tumors) followed by postoperative radiotherapy (50-70 Gy, average 59.2 Gy). No recurrences of MLS were observed locally at the mean follow-up of 58.1 months. The development of severe radiation-related complications was not detected. This medium follow-up study suggested that this method can achieve a high rate of local control.

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：BRITISH MED JOURNAL PUBL GROUP  

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：WILEY-BLACKWELL  

Recombinant adenovirus is used as a competent vector in a wide spectrum of cancer gene therapies. Adenovirus infection depends on coxsackievirus and adenovirus receptor (CAR)-mediated virus attachment to the cell surface. However, the expression levels of CAR and the efficiency of adenoviral gene transduction in musculoskeletal tumors have not been systematically investigated. To study the feasibility of gene therapy in musculoskeletal tumors, the expression levels of CAR and the antiproliferative effect of an adenovirally transduced wild-type p53 tumor suppressor gene were examined in 15 distinct musculoskeletal tumor cell lines, 19 tumor tissue samples, and the corresponding pathologically unremarkable mesenchymal tissues. The expression levels of the CAR gene were significantly higher in six of seven osteosarcoma cell lines and two of five osteosarcoma tissue samples than in the other cell lines, musculoskeletal tumors, and mesenchymal tissues. CAR expression levels were closely correlated with adenoviral gene transduction efficiency and the antiproliferative effect of a transduced adenoviral p53 gene in the tested cell lines. In addition, an immunocytochemical study confirmed that transfected green fluorescent protein (GFP) borne by Ad-CAG-GFP was expressed at the cell surface of CAR-positive cells. These results indicate that CAR expression is a critical determinant of transduction efficiency in adenovirus-based gene therapy. Most osteosarcomas appeared to express high levels of CAR, and thus adenovirus-mediated p53 gene therapy is likely to be suitable for the treatment of such tumors.

DOI： 10.1111/j.1349-7006.2003.tb01354.x

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：Springer Japan  

The effect of dexamethasone, a synthetic glucocorticoid, on in vitro and in vivo growth and differentiation of the human chondrosarcoma cell line (OUMS-27) was studied. Cells were treated with various doses of dexamethasone, and increasing doses produced an inhibitory effect on OUMS-27 tumor cell proliferation and induced maturation. Cell counts for OUMS-27 on day 9 ranged from 59% of the control at 10-8 M to 45% of the control at 10-5 M dexamethasone. Northern blot analysis revealed that the type II collagen mRNA level in cells given dexamethasone was lower than that in the controls, and the type X collagen mRNA level was higher than that in the controls. Phase-contrast microscopy revealed that cells grown in control medium formed monolayers consisting of small, polygonal cells, whereas dexamethasone-treated cells became larger and more irregular in shape. In the in vivo study the growth rate of masses in nude mice indused by inoculating OUMS-27 cells was also reduced in a dosedependent manner with dexamethasone administration. These results suggest that dexamethasone caused growth inhibition and induced chondrogenic maturation of human chondrosarcoma cells.

DOI： 10.1007/s10776-003-0650-y

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：KARGER  

Objective: The clinical features and the management of alveolar soft part sarcoma (ASPS) are not well known. The efficacy of chemotherapy for soft tissue sarcoma, including high-dose ifosfamide and cisplatin, has not been established yet. Some reports suggest ASPS may occur primarily in bone. Methods: We report on a series of 57 patients with ASPS over 20 years. Their ages ranged from 7 to 75 years (mean 25). Results:There were 37 females and 20 males. Thirteen lesions (23%) showed bone involvement at the primary site, and 6 of them were diagnosed as bone tumors at presentation. Thirty-seven patients had distant metastases at presentation. Tumor size, bone involvement at the primary site and the presence of metastases at presentation were prognostic indicators (p < 0.05). Marginal exicision with radiotherapy or wide excision without radiotherapy achieved good local control. Chemotherapy was performed in 47 patients with different regimens. Two patients treated with intraarterial chemotherapy regimens responded partially, but intravenous chemotherapy with high-dose ifosfamide or cisplatin failed. Conclusions: ASPS can present primarily as a bone tumor. No advantage of chemotherapy with high-dose ifosfamide or cisplatin could be demonstrated. Copyright (C) 2003 S. Karger AG, Basel.

DOI： 10.1159/000071199

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記述言語：日本語   出版者・発行元：東北整形災害外科学会  

上肢悪性腫瘍に対する三角筋筋膜皮弁を用いた肩甲帯離断術を行った3例を経験した.症例1は61歳男性.右環指爪下部原発の悪性黒色腫で,腫瘍は尺骨,正中,筋皮神経内に浸潤していた.リンパ節以外の転移を認めず,三角筋筋膜皮弁で断端被覆を行う肩甲帯離断術を施行した.しかし,術後8年後に後腹膜腔への転移で死亡した.症例2は54歳男性.小児期の前腕部火傷瘢痕部に生じた扁平上皮癌で,前医に広範切除・脇窩リンパ節郭清を施行されたが,腋窩部で再発を来して受診した.症例1と同様に腋窩部切除の皮切デザインで手術を行ったが,術後約11ヵ月後に多発肺転移にて死亡した.症例3は76歳女性.乳癌切除後の左上肢リンパ血管肉腫で,左上腕内側の紫斑が上肢全体に拡大し,潰瘍も形成され受診した.腫瘍は上肢全体に進展し,左上背部,左前胸部までの浸潤を疑い,本術式を行った.術後約11ヵ月経過で再発,転移はない

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記述言語：日本語   出版者・発行元：東北整形災害外科学会  

足部に原発し悪性化した多発性内軟骨腫症の2例を経験した.症例1は初診時17歳男性.左足部痛が持続,内軟骨腫症の診断で左脛骨遠位部,母趾基節に掻爬骨移植施行した.31歳時に脛骨に再発し,舟状骨の腫瘍出現で掻爬骨移植を施行したが,32歳時に舟状骨部の疼痛出現,X線,CTで舟状骨外への浸潤を認め,悪性化と判断して左下腿切断を施行した.術後9年経過で再発,転移はない.症例2は初診時52歳男性で,左足部痛で受診し,X線で左第4,5趾,第5中足骨,踵骨,立方骨に透亮像を認め,多発性内軟骨腫症の診断で第4,5趾はMTP関節離断,第5中足骨と踵骨は掻爬骨移植を行った.組織診断は内軟骨腫であった.その後,69歳時に左足部の持続疼痛出現し,X線で立方骨の透亮像増大,骨皮骨の破壊と踵骨再発を認め,左下腿切断を施行した.術後半年経過で再発,転移はない.両症例とも下腿切断時の組織診断は軟骨肉腫であった

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：LIPPINCOTT WILLIAMS & WILKINS  

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：COMPANY OF BIOLOGISTS LTD  

The periodontal ligament (PDL) is a connective tissue located between the cementum of teeth and the alveolar bone of the mandibula. It plays an integral role in the maintenance and regeneration of periodontal tissue. The cells responsible for maintaining this tissue are thought to be fibroblasts, which can be either multipotent or composed of heterogenous cell populations. However, as no established cell lines from the PDL are available, it is difficult to assess what type of cell promotes all of these functions. As a first step to circumvent this problem, we have cloned and characterized cell lines from the PDL from mice harboring a temperature-sensitive SV 40 large T-antigen gene. RT-PCR and in situ hybridization studies demonstrated that a cell line, designated PDL-L2, mimics the gene expression of the PDL in vivo: it expresses genes such as alkaline phosphatase, type I collagen, periostin, runt-related transcription factor-2 (Runx2) and EGF receptor, but does not express genes such as bone sialoprotein and osteocalcin. Unlike osteoblastic cells and a mixed cell population from the PDL, PDL-L2 cells do not produce mineralized nodules in the minearlization medium. When PDL-L2 cells were incubated in the presence of recombinant human bone morphogenetic protein-2 alkaline phosphatase activity increased and mineralized nodules were eventually produced, although the extent of mineralization is much less than that in osteoblastic MC3T3-E1 cells. Furthermore, PDL-L2 cells appeared to have a regulatory mechanism by which the function of Runx2 is normally suppressed.

DOI： 10.1242/jcs.00098

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：JOHN WILEY & SONS INC  

Prominent osteoconductive activity and the biodegradable nature of beta tricalcium phosphate (beta-TCP) for bone grafts in animal experiments has been reported. A new type of beta-TCP has been manufactured at extraordinarily high purity and has been available as potent bone grafting substitute for clinical use. The histological features of grafted beta-TCP in human bone have been analyzed. A 33-year-old female with a bone tumor of the proximal femur underwent curettage and beta-TCP graft under the diagnosis of probable benign fibrous dysplasia. Four weeks later, the proximal femur, including the grafted beta-TCP was resected because of the final diagnosis of the curettaged materials was osteosarcoma. The resected specimen revealed abundant direct new bone apposition on beta-TCP. There was no cartilaginous tissue or enchondral ossification. Bone formation was more prominent in the periphery of the grafted area than in the center. There was a considerable number of osteoclast-like giant cells surrounding the beta-TCP. This case illustrated that highly purified beta-TCP had prominent osteoconductive activity and biodegradable nature in human bone. (C) 2002 Wiley Periodicals, Inc.

DOI： 10.1002/jbm.10380

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記述言語：日本語   出版者・発行元：東北整形災害外科学会  

21歳女の第1子である女児.妊娠25週,妊婦検診時の超音波検査で左下腿腫瘤を指摘された.妊娠29週より心不全徴候が出現したため経母体的投与によるジギタリス療法を開始し,妊娠31週,胎児仮死のため帝王切開で出生した.出生時,左下肢に径15×16cmの巨大な充実性腫瘤を認めた.腫瘍への血流の供給によると思われる心不全が出現した.針生検を行い,左下肢腫瘤を線維肉腫と診断した.心不全に加え,腫瘍そのものの増大と腫瘍上の皮膚の壊死傾向を認め,全身状態のすみやかな改善が必要と判断し,大腿切断術を施行した.術後よりすみやかに心不全は改善した.現在,術後112日の時点で局所再発,遠隔転移を疑わせる所見は認めていない

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記述言語：日本語   出版者・発行元：(一社)日本皮膚悪性腫瘍学会  

75歳男.左母指背側に隆起性腫瘤が出現した.左母指DIP関節背側の3.5×3.2cm大の暗紫紅色の腫瘤は,類円形で表面平滑,一部血痂を付着し,弾性軟,脆弱で易出血性,疼痛を伴っていた.胸部X線,CTで右上肺葉に径10cmを越える腫瘤陰影を認めた.強い疼痛と腫瘤の急速な増大で左母指切断術を施行した.病理組織では,腫瘍は核の濃染した小型な類円形細胞を主体とし,一部管腔様構造も認めた.また,組織の一部に腺腔様構造を取り囲む様に,または数珠状に配列する大型の上皮様細胞や,柵状に配列する紡錘形細胞を認め,二相性構造であった.免疫組織では,小型および紡錘形細胞はビメンチン陽性,上皮様細胞はCAM5.2陽性で,左母指の腫瘤は低分化型滑膜肉腫と診断し,肺腫瘤はその転移と考えた.全身状態より手術適応がないと判断し,肺腫瘤には放射線療法を開始したが,発症後6ヵ月で呼吸不全のため永眠した

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記述言語：英語   掲載種別：研究論文（学術雑誌）  

DOI： 10.1016/S0167-4781(01)00232-9

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その他リンク： http://orcid.org/0000-0003-4698-460X

記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：INT INST ANTICANCER RESEARCH  

Background: Clear cell chondrosarcoma is a rate bone tumor, which is sometimes misdiagnosed as a different bone neoplasm. Materials and Methods: The files of 6 patients with clear cell chondrosarcomas were reviewed. Histological slides, radiographic studies, and pre- and post-operative serum alkaline phosphatase (ALP) levels were evaluated. Molecular and histochemical analyses of ALP were documented in one case of clear cell chondrosarcoma. Results: Pre-operative serum ALP levels were elevated in 3 patients, and were normal in another 3 patients. After removal of the tumors, the enzyme levels decreased in all patients and returned to normal in 3 patients, who had pre-operative high ALP levels. Enzyme histochemical and molecular analyses demonstrated that the tumor produced ALP. Conclusion: Clear cell chondrosarcoma produces ALP, which can be used as a tumor marker in diagnosis and follow-up.

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記述言語：英語   掲載種別：研究論文（学術雑誌）   出版者・発行元：Springer Japan  

Circulating tumor cells in the blood play a central role in the metastatic process. There have been no reports describing the relationship between lung metastasis and circulating osteosarcoma cells. We developed a system with a polymerase chain reaction assay based on an enzyme-linked immunosorbent assay (PCR-ELISA) to detect circulating osteosarcoma cells in a mouse metastatic model. Osf2/Cbfal, hereafter called Osf2, a member of the runt family of transcription factors, was used as a target gene. One splicing variant of Osf2 mRNA was identified and its expression was restricted to the bones and osteosarcomas. The amount of the splicing variant of Osf2 mRNA was significantly higher in the blood of mice with metastasis than in the blood of the control group. The PCR-ELISA using Osf2 mRNA is a potential method to detect circulating osteosarcoma cells in peripheral blood.

DOI： 10.1007/s007760100046

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記述言語：日本語   出版者・発行元：新潟整形外科研究会  

症例は76歳の男性で主訴は多関節痛,両下腿紅斑,腫脹である.入院時,MRI所見は左膝T1強調像では脛骨で虫食い状の高低輝度混在,T2強調像でも同様で,一部低輝度に変化した部分が認められた.また右手のMRI T1強調像では,中指,環指,小指に低輝度陰影がみられた.入院時,アミラーゼ,リパーゼが異常高値で非定型的膵炎と考えメチル酸ガベキサート(FOY)を投与し,関節痛は膵炎合併骨内脂肪壊死と判断した.その後FOYによる膵炎治療の結果徐徐に疼痛が軽減し,アミラーゼ,リパーゼの正常化と共に疼痛消滅,独歩可能となった

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記述言語：日本語   出版者・発行元：新潟整形外科研究会  

30歳女.嚢腫を基部で切離し,嚢腫壁を縫縮することにより良好な結果を得た

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記述言語：日本語   出版者・発行元：東北整形災害外科学会  

坐骨腫瘍3例(外骨腫,血管腫,動脈瘤様骨嚢腫),恥骨腫瘍6例(軟骨肉腫3例,骨肉腫1例,Ewing肉腫1例,外骨腫1例)の治療成績を報告した.いずれも局所再発は無く,機能も極めて良好であった.Ewing肉腫の1例のみ腫瘍死したが,他の悪性腫瘍4例は無病生存していた.恥骨の一期的再建は不要と思われた

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記述言語：日本語   出版者・発行元：東北整形災害外科学会  

1)片麻痺を合併した大腿骨頸部骨折症例について検討した. 2)臨床像としては,麻痺側の骨折率は80%と高く,性別では健常群に比べ男性の割合が高いという特徴があった. 3)麻痺合併症例は入院日数が長期化する傾向があり,退院時歩行再獲得率も低いことから,リハビリ病院等の中間施設の整備の必要性が示唆された

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記述言語：日本語   出版者・発行元：東北整形災害外科学会  

1)外傷性骨折後偽関節に対し,手術治療を行った20例を検討した. 2)初回偽関節手術で骨癒合が得られたのは,人工骨頭置換術2例を除く18例中15例. 3)偽関節手術として,骨移植+圧迫プレート法は,確実性のある術式である. 4)感染を伴わない難治性の偽関節に対しては,血管柄付き腓骨移植を行うことにより,ほぼ良好な経過を得た

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記述言語：日本語   出版者・発行元：東北整形災害外科学会  

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記述言語：日本語   出版者・発行元：新潟整形外科研究会  

1)性別では麻痺側群は男性の割合が高いのに対し,健側群は全例女性であり有意差を認めた.この要因の一つとして,それぞれ麻痺側の易骨折性の関与,骨粗鬆症の関与が考えられた. 2)麻痺側群は歩行能力再獲得率に差はないものの,入院日数が長期化する傾向があった.これは麻痺と同一側に骨折が加わることにより,歩行機能を回復する迄に長い期間を要したためと考えられた

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：東北整形災害外科学会  

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記述言語：日本語   出版者・発行元：東北整形災害外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(株)南江堂  

＜文献概要＞はじめに 運動器に発生する腫瘍は,一般に骨・軟部腫瘍と称され,骨腫瘍は骨から発生し,軟部腫瘍は軟部組織から発生する腫瘍の総称である.全身のあらゆる部位に発生し,骨・軟骨・皮下・筋肉・脂肪・血管・腱・滑膜などに由来する間葉系腫瘍に末梢神経系腫瘍を加えたものと考えることができる.しかし,由来細胞がはっきりしない腫瘍も含まれ,これらの病理組織学的な分類は非常に多岐にわたる.腫瘍性疾患の診断は最終的には病理診断に頼ることになるが,診断する多くの病理医にとっても経験することが少ない疾患が多く,整形外科医との協働は重要である.そこで本稿では,小児の運動器に発生し,日常診療で遭遇する頻度の高い疾患を対象として解説する.

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：東日本整形災害外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

J-GLOBAL

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：東北整形災害外科学会  

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記述言語：日本語   出版者・発行元：東北整形災害外科学会  

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記述言語：日本語   出版者・発行元：東北整形災害外科学会  

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記述言語：日本語   出版者・発行元：(NPO)日本緩和医療学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：新潟整形外科研究会  

症例は6歳男児で、2歳時に多発性軟骨性外骨腫症と診断され、自覚症状なく経過観察した。3ヵ月前に家族が歩行時の躓きと右下垂足に気づいた。X線検査にて右腓骨頭部に骨軟骨腫増大を認めた。単純X線で右腓骨頭に骨髄腔と連続した腫瘤を認め、初診時と比較し増大していた。右脛腓骨遠位にも腫瘤を認めたが、足関節の内外反変形は認めなかった。単純CTで右腓骨頭前方外側に隆起性病変を認め、MRI造影T1強調脂肪抑制像およびT2強調脂肪抑制像で同部位に骨と同信号の腫瘤を認め、T2強調脂肪抑制像で腫瘤辺縁に軟骨帽と思われる約2mmの高信号域を認めた。右下垂足の原因として腓骨神経領域に一致した筋力低下と右腓骨頭部の骨軟骨腫増大を認めたことより、腓骨頭骨軟骨腫による腓骨神経圧迫を疑い、手術を施行した。腫瘍上に神経が多数分枝し、腫瘍の一塊での摘出は囲難と考え、断片的に腫瘤を摘出して神経の圧迫を解除した。腫瘤の病理診断は、骨軟骨腫であった。術後2ヵ月迄に下肢筋力は徐々に回復し、術後4ヵ月で走行も可能となった。

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記述言語：日本語   出版者・発行元：(株)全日本病院出版会  

軟部腫瘍の診断は、問診、視診、触診といった基本的な診察を行ったあとに、MRIを中心とした画像検査を行い、最終的には組織生検により病理学的な診断を確定していく。しかし、疾患そのものに多様性があり、希少なものが多いため、これらの検査を行っても特徴的な所見が得られず、診断に難渋することもしばしばである。一方で、日常診療で目にすることが多い、脂肪腫やガングリオン、類上皮嚢胞(粉瘤)、神経鞘腫などにおいては、比較的特徴的な所見がみられる。よって、これらの良性腫瘍を診断する際に、自信を持ってしっかりと診断できることが重要であり、そうでないものは常に悪性の可能性を考慮して、早めに専門医へ相談するという姿勢が必要だろう。本稿では、良性軟部腫瘍診断においてピットフォールに陥らないための基本的なポイントを中心に解説する。(著者抄録)

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(NPO)日本緩和医療学会  

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記述言語：日本語   出版者・発行元：東北整形災害外科学会  

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記述言語：日本語   出版者・発行元：東北整形災害外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(株)全日本病院出版会  

膝痛は、極めて頻度の高い病態であるが、その原因は多岐にわたる。整形外科医が得意とする骨関節の損傷、変性、炎症による疾患だけでなく、血管性疾患や悪性疾患による膝痛、あるいは全身疾患の随伴症状の一つとして膝痛が生じることもある。このような疾患では膝の局所所見のみならず、全身的な病変や症状の確認、詳細な病歴の聴取が診断上の重要な手掛かりになる。本稿では膝痛をきたす疾患のうち、日常診療で見落としがちな「整形外科疾患以外の疾患」について取り挙げ、疾患の特徴について概説する。(著者抄録)

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：新潟整形外科研究会  

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記述言語：日本語   出版者・発行元：新潟整形外科研究会  

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記述言語：日本語   出版者・発行元：新潟整形外科研究会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：東日本整形災害外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：東北整形災害外科学会  

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記述言語：日本語   出版者・発行元：(NPO)日本緩和医療学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：(公社)日本整形外科学会  

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記述言語：日本語   出版者・発行元：新潟医学会  

CiNii Article

CiNii Books

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その他リンク： http://search.jamas.or.jp/link/ui/2016151272

記述言語：日本語   出版者・発行元：(株)全日本病院出版会  

「しこり・こぶ・腫れ」を主訴に医療機関を受診する患者さんは少なくないが、その患者さんを目の前にして、どんな疾患を鑑別診断として考えるべきか判断することは容易でない。筆者の印象では、患者さんは「脂肪の塊」という表現で軟部腫瘤を示すことも多いようだが、実際にそれが脂肪腫であるケースは必ずしも多くなく、なかには悪性軟部腫瘍(軟部肉腫)のような、見逃してはいけない疾患も含まれる。診断を進めるうえで、まず初めに行うのは問診、視診そして触診といった診察であるが、ここで得られる年齢や発生部位、疼痛の有無、大きさの変化などの情報は、診断の鑑別を進めるうえで非常に重要なポイントとなる。血液生化学検査はすべての軟部腫瘤の診断に必ずしも必要な検査ではないが、疾患の鑑別のみならず、時に病勢を判断するためにも重要な情報が含まれる場合もある。本稿では、軟部腫瘤診療における臨床所見の取り方とその解釈、血液生化学データの要点について解説する。(著者抄録)

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記述言語：日本語   出版者・発行元：(株)メジカルビュー社  

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記述言語：日本語   出版者・発行元：新潟整形外科研究会  

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記述言語：日本語   出版者・発行元：(株)南江堂  

腫瘍用人工膝関節全置換術(TKA)後の膝蓋骨の位置異常と患肢機能について検討した。大腿骨遠位部骨腫瘍に対して腫瘍用TKAを施行し、2年以上経過しX線像で膝蓋骨高位および亜脱臼位(傾斜角)の評価が可能であった23例を対象とした。膝蓋骨低位群8例、正常群15例で、平均膝屈曲角度は低位群が71.3°で、正常群の107.3°に比べて有意に小さかった。ISRと膝属曲角度には中等度の相関関係を認めた。平均ISOLSスコアは両群で有意差はなかった。膝蓋骨傾斜角亜脱臼群6、正常群17例で、平均膝関節屈曲角は亜脱臼群が76.7°で、正常群の101.2°に比べて小さい傾向があった。平均ISOLSスコアは両群で有意差はなかった。ISRと傾斜角には、中等度の負の相関関係を認め、膝蓋骨低位では傾斜角が大きくなる傾向があった。

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記述言語：日本語   出版者・発行元：(株)メジカルビュー社  

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記述言語：日本語   出版者・発行元：金原出版(株)  

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記述言語：日本語   出版者・発行元：新潟医学会  

本院はがん対策基本法の施行に伴い、平成19年にがん診療拠点病院に指定され、その整備指針に従って、通院治療室・緩和ケア室・がん相談支援室・がん登録室の4部門からなる腫瘍センターが開設された。同時に、地域連携機能の強化と緩和医療に関する医療従事者の意識向上と知識の普及・啓発なども進められてきた。そこで、これまでの緩和ケアチームと地域保健医療推進部の実績を振り返り、これから期待される活動の方向性について考察した。(著者抄録)

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記述言語：日本語   出版者・発行元：(公社)日本皮膚科学会  

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記述言語：日本語   出版者・発行元：(公社)日本皮膚科学会  

J-GLOBAL

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記述言語：日本語   出版者・発行元：(公社)日本皮膚科学会  

J-GLOBAL

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記述言語：日本語   出版者・発行元：(株)南江堂  

インフォームド・コンセントの得られた骨移植を必要とする2例から2mlの骨髄を腸骨から採取し、高純度β-リン酸三カルシウム(β-TCP)ディスク上に培養した。これを7〜9週齢のsevere combined immune deficiency(SCID)マウスの背部筋層内に移植し、9週後にディスクを採取して4%パラホルムアルデヒドにより固定し、EDTA脱灰を行った後にパラフィン包埋切片を用いて組織解析を行った。更に、以前に報告した方法に準じて連続切片を用いてヒト特異的Aluプローブとマウス特異的L1プローブを用いてDNA in situ hybridizationを行った。その結果、移植後9週で2例ともβ-TCP上に直接骨形成を認めた。ヒト特異的Aluプローブ陽性細胞はβ-TCP新生骨の境界部にのみ僅かに認め、マウス特異的L1は陰性であった。また、骨内に存在する骨細胞は、マウス特異的L1プローブに陽性かつAlu陰性であった。その他、骨周囲の血管内皮細胞や線維芽細胞の紡錘型細胞は全てL1陽性、Alu陰性で、マウス由来細胞と考えられた。一方、対照として細胞を添加しなかったβ-TCPには骨形成は認めなかった。

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記述言語：日本語   出版者・発行元：(株)南江堂  

骨・軟部腫瘍に対する穿刺吸引細胞診の有用性について検討した。骨腫瘍は178例の穿刺例のうち細胞が採取できなかった不適切材料が21例で、穿刺のみで最終診断がなされなかったもの48例にみられた。通常型の骨肉腫、Ewing肉腫、癌転移、リンパ腫では100%正診であった。感度、特異度偽陽性率、偽陰性率はそれぞれ95.5%、100%、0%、4.5%で、正診率は97.2%であった。誤診例は非定型的骨肉腫1例とgrade Iの軟骨肉腫が2例で、良性と判定された。軟部腫瘍は826例に対して穿刺が行われたが、不適切材料が109例あり、穿刺のみで最終診断がなされなかったものが394例にみられた。感度、特異度、偽陽性率、偽陰性率はそれぞれ85.5%、96、9%、2.5%、14.5%であり、正診率は91.3%であった。MFH、粘液型脂肪肉腫、横紋筋肉腫、リンパ腫、癌転移などはほぼ100%正診であった。

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J-GLOBAL

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記述言語：英語   掲載種別：研究発表ペーパー・要旨（国際会議）   出版者・発行元：AMER ASSOC CANCER RESEARCH  

Web of Science

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記述言語：日本語   出版者・発行元：(一社)日本皮膚悪性腫瘍学会  

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記述言語：日本語   出版者・発行元：(株)全日本病院出版会  

血管腫の血管は壁に筋層を持たない毛細血管の構造のため、剥離操作で損傷されやすく、電気凝固に対しても抵抗性である。したがって、電気メスでは十分な止血が得られず、特に部分切除(腫瘍内切除)では大出血をきたす場合があった。我々は筋肉内血管腫に対する新しい手術手技を開発し、周囲二重結紮法(circumferential parallel ligation method;CPL法)と名づけた。CPL法は切除線の両側を針糸を用いて平行に結紮した後、結紮部の間を切開する方法である。針糸結紮は盲目的に行い、深部に向かって結紮を繰り返すことで、最終的に血管腫を腫瘍内で切除できる。従来法とCPL法で比較したところ、CPL法で有意に出血量が少なく、CPL法では全例輸血を必要としなかった。CPL法は筋肉内血管腫の切除において確実に出血がコントロールできる有用な方法である。(著者抄録)

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記述言語：日本語   出版者・発行元：(株)医学書院  

良性骨腫瘍そう爬術後に人工骨移植を行うことで優れた臨床成績が報告されている.現在わが国で広く使用されている人工骨には,ハイドロキシアパタイト,骨ペースト,β-リン酸三カルシウム(β-TCP)などがある.各材料,さらには同じ材料でもメーカーや気孔率,気孔の連通性,表面形状によりその特性が異なり,自分の使用する材料の特性をよく知ることが重要である.筆者が好んで用いているβ-TCPは初期強度は弱いが吸収置換され,旺盛な骨伝導能をもつ優れた材料であり,骨腫瘍領域において優れた臨床成績の報告が多い.(著者抄録)

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記述言語：日本語   出版者・発行元：整形外科バイオマテリアル研究会  

鍍銀染色法を用いてこの高純度β-リン酸三カルシウム(β-TCP)のマイクロポア構造内にコラーゲンの形成がみられることを報告した。今回、免疫染色と電子顕微鏡的観察を加え更なる解析を行った。8週齢のFisherラット大腿骨に小孔を作成し気孔率75%、100〜500μmのマクロポアと5μm未満マイクロポアを有するβ-TCP顆粒を移植した。ラット移植後4日ではまだ骨形成は確認されないが早期にマイクロポア内に鍍銀染色陽性所見がみられ、この線維はβ-TCP外のコラーゲン線維と一部直接結合した。免疫染色ではTCP間隙、マクロポア内の未分化な紡錘形細胞、細胞外基質、およびβ-TCPの内部表層に1型コラーゲンの陽性所見がみられた。移植後7日でβ-TCP表面には未熟骨が形成され、骨から連続するようにコラーゲン線維の形成を確認した。

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記述言語：日本語   出版者・発行元：金原出版(株)  

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記述言語：日本語  

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記述言語：日本語   出版者・発行元：(株)南江堂  

骨腫瘍手術に際して生じた骨欠損に対する高純度β-リン酸三カルシウム(β-TCP)移植の臨床成績を検討した.移植後,3ヵ月以上の経過観察が可能であった42例を対象とした.全例で移植β-TCP周囲に速やかに自家骨の形成が認められ,術後感染やアレルギー反応といった有害事象はみられなかった.2例で組織学的解析を行ったところ,術後2週では未分化な細胞の付着,小孔内への血管の新生像,破骨細胞様の巨細胞の付着がみられた.術後4週では盛んな骨形成がβ-TCPを足場として始まっており,同時に破骨細胞様巨細胞による吸収像がみられた.β-TCPは骨腫瘍手術における骨補填材料として優れた性質を持っており,移植後早期から盛んな骨新生がみられることが判明した

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記述言語：英語   掲載種別：速報，短報，研究ノート等（学術雑誌）   出版者・発行元：AMER ASSOC CANCER RESEARCH  

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記述言語：日本語   出版者・発行元：新潟大学  

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その他リンク： http://search.jamas.or.jp/link/ui/2005034670

記述言語：日本語   出版者・発行元：歯科基礎医学会  

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記述言語：日本語   出版者・発行元：(株)アークメディア  

1994〜2002年まで当科で悪性骨軟部腫瘍切除後の骨欠損に対して行った照射骨移植20例(うち,女性12例.9〜62歳,平均年齢35.3歳.骨腫瘍15例,軟部腫瘍5例)の治療成績を検討した.その結果,照射骨からの再発はなく,骨癒合率も85%と良好であった.術後合併症については,骨幹部の欠損に対する移植,及び一部の皮質骨のみ切除し照射する術式では発症しなかったが,関節軟骨も含めて照射しそのまま戻す,いわゆる骨関節移植(以下,OAG)で関節面の陥没や感染,関節破壊が認められた.とくに下肢への移植では5例全てに関節面の陥没や関節破壊が認められた.OAGでは,長期的関節機能の温存が困難であるため,二次的再建の必要性を視野に入れるべきであると考えられた

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記述言語：日本語   出版者・発行元：(株)アークメディア  

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記述言語：日本語   出版者・発行元：(株)医学書院  

骨軟部肉腫535例(平均42歳).軟部肉腫303例,骨原発肉腫232例であった.術後2〜294ヵ月で,転移は213例に生じ,肺189例,骨63例,軟部38例,脳23例,肝23例,後腹膜腔12例等であった.稀な転移部位としては腸管6例,脊椎硬膜外腔3例,膵臓,心臓,腎臓各2例,脊髄,喉頭各1例があった.脂肪肉腫65例を亜型でみると,高分化型では転移例がなかったが,粘液型・円形細胞型では30例中10例に転移を認め,肺転移3例,骨転移7例,軟部転移10例,脳転移1例,肝転移5例,後腹膜腔転移3例であった.組織型と転移部位で関連性が高かったのは,粘液型・円形細胞型の他にはMFHの腸管転移,胞巣状軟部肉腫の骨・脳転移,骨ユーイング肉腫の骨転移等があった.転移発見後の平均余命は,肺16ヵ月,骨10ヵ月,軟部18ヵ月,脳5ヵ月,肝6ヵ月と不良であった

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記述言語：英語   掲載種別：研究発表ペーパー・要旨（国際会議）   出版者・発行元：ELSEVIER SCIENCE INC  

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記述言語：英語   掲載種別：研究発表ペーパー・要旨（国際会議）   出版者・発行元：ELSEVIER SCIENCE INC  

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記述言語：日本語   出版者・発行元：(株)南江堂  

骨・軟部腫瘍571例639病変(男287例,女284例).骨腫瘍は202病変のうち172病変で診断に適する細胞が採取され,軟部腫瘍は437病変中392病変であった.成績は骨腫瘍で感度97.6%,特異度94.4%,偽陽性率5.6%,偽陰性率2.4%,正診率96.5%,軟部腫瘍は各々93.0%,93.5%,6.5%,7.0%,93.4%であった.骨腫瘍の偽陽性は5例で,骨巨細胞腫(GCT),疲労骨折と骨折を伴うGCT,myofibromatosis,腸骨の慢性骨髄炎が各1例であった.偽陰性は2例で,低悪性骨肉腫と甲状腺癌の腸骨転移であった.軟部腫瘍の偽陽性は20例で,脂肪腫,神経鞘腫,血管腫,破裂したガングリオン等であった.脂肪腫は35例中7例,神経鞘腫は25例中5例,血管腫は15例中3例が偽陽性となった.偽陰性は悪性神経鞘腫3例,分化型脂肪肉腫,線維肉腫,胞巣状軟部肉腫が各1例であった.骨腫瘍中の癌転移の正診率は97.3%で,軟部腫瘍の癌転移,骨・軟部の悪性リンパ腫と白血病は100%であった

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記述言語：英語   掲載種別：速報，短報，研究ノート等（学術雑誌）   出版者・発行元：OXFORD UNIV PRESS  

DOI： 10.1093/rheumatology/keg086

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記述言語：英語   掲載種別：研究発表ペーパー・要旨（国際会議）   出版者・発行元：AMER SOC CELL BIOLOGY  

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記述言語：日本語   出版者・発行元：新潟県医師会  

骨肉腫細胞株4株を含む骨軟部悪性腫瘍細胞株10株と,腫瘍組織26検体,正常間葉系組織を用いて,アデノウイルスレセプターの発現について調べ,p53アデノウイルスベクターによる遺伝子治療の可能性について検討した.その結果,骨軟部悪性腫瘍の中では,骨肉腫において特にアデノウイルスレセプターの発現が高く,また正常の間葉系組織では低いことから,アデノウイルスベクターを骨肉腫の腫瘍組織内へ直接投与した際に,周囲の正常組織への感染による影響の少ない効率的な遺伝子治療が行えるものと考えられた

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記述言語：日本語   出版者・発行元：(株)医学書院  

軟部肉腫37例に対し,組織型に拘わらず,術後照射を併用し,軟部肉腫の切除縁が縮小可能かどうか検討した.対象の経過観察期間は平均43.7ヵ月であり,照射線量は平均59.4Gy,すべて辺縁切除,または腫瘍内切除であった.再発率は8.1%,累積5年生存率は67.7%であった.合併症は43.2%と高率に認められ,関節拘縮が48.1%と多かった.このため,Ennekingの評価法による患肢機能は88.9%となった.関節拘縮が一番大きな問題であったが,術後照射は切除縁を縮小する有効な補助療法であると思われた

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記述言語：日本語   出版者・発行元：(株)医学書院  

多発性神経鞘腫瘍の分類における鑑別点と,悪性末梢神経鞘腫瘍(MPNST)の発生について検討した.過去30年間の多発性神経鞘腫瘍115例のうち,National Institutes of Healthの診断基準によると,neurofibromatosis(NF)1が78例,NF2が7例,その他schwannomatosisが15例,segmental NFが2例,データ不足で分類不能が13例であり,各々の混合型はみられなかった.MPNSTはNF1例のみに14例発生していた.鑑別点としては,腋窩小色素斑,虹彩過誤腫がNF1に特異的で重要であり,カフェオレ斑はNF1以外の病型にもしばしばみられる.NF1以外にはMPNSTはほとんど発生しないこから,これらの病型を臨床的に判別する事は重要である

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記述言語：日本語  

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記述言語：英語   掲載種別：研究発表ペーパー・要旨（国際会議）   出版者・発行元：ELSEVIER SCIENCE INC  

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症例は6歳男児で、2歳時に多発性軟骨性外骨腫症と診断され、自覚症状なく経過観察した。3ヵ月前に家族が歩行時の躓きと右下垂足に気づいた。X線検査にて右腓骨頭部に骨軟骨腫増大を認めた。単純X線で右腓骨頭に骨髄腔と連続した腫瘤を認め、初診時と比較し増大していた。右脛腓骨遠位にも腫瘤を認めたが、足関節の内外反変形は認めなかった。単純CTで右腓骨頭前方外側に隆起性病変を認め、MRI造影T1強調脂肪抑制像およびT2強調脂肪抑制像で同部位に骨と同信号の腫瘤を認め、T2強調脂肪抑制像で腫瘤辺縁に軟骨帽と思われる約2mmの高信号域を認めた。右下垂足の原因として腓骨神経領域に一致した筋力低下と右腓骨頭部の骨軟骨腫増大を認めたことより、腓骨頭骨軟骨腫による腓骨神経圧迫を疑い、手術を施行した。腫瘍上に神経が多数分枝し、腫瘍の一塊での摘出は囲難と考え、断片的に腫瘤を摘出して神経の圧迫を解除した。腫瘤の病理診断は、骨軟骨腫であった。術後2ヵ月迄に下肢筋力は徐々に回復し、術後4ヵ月で走行も可能となった。

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