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

Background: Scapulectomy requires not only joint resection but also wide resection of the shoulder girdle muscles. Even the significance of reconstruction has not yet been determined because of the difficulties in comparing the different conditions. The purpose of this study was to investigate factors that influence functional outcomes after scapulectomy in a multicenter study.
Methods: This retrospective study comprised 48 patients who underwent total or subtotal scapulectomy and were followed for at least one year after surgery. Patients were registered at the Japanese Musculoskeletal Oncology Group affiliated hospitals. Soft tissue reconstruction for joint stabilization was performed when there was enough remaining tissue for reconstruction of the rotator cuff and tendons. In 23 cases, humeral suspension was performed. The average follow-up period was 61.9 months. Multivariate analysis was performed using the patient's background to determine which factors influence the Enneking functional score or active range of motion.
Results: The average functional score was 21.1 out of 30. Active shoulder range of motion was 42.7 degree in flexion, 39.7 degree in abduction, 49.6 degree of internal rotation and 16.8 degree of external rotation. The amount of remaining bone influenced functional outcome, which means that preserving the glenoid or the acromion lead to better function compared to total scapulectomy (p<0.01). Factors that influenced each functional measure include the amount of remaining bone, soft tissue reconstruction, the length of the resected humerus and nerve resection (p<0.05).
Conclusion: Although shoulder function was almost eliminated following total or subtotal scapulectomy, minimal resection of bone, and soft tissue reconstruction should lead to better function.

DOI： 10.1371/journal.pone.0100119

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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: Cyclooxygenase-2 (COX-2) is a key enzyme in the conversion of arachidonic acid to prostanoids, and its activation is associated with carcinogenesis as well as inflammation. The antitumor effect of selective COX-2 inhibitors has been noted in various malignancies. Malignant peripheral nerve sheath tumor (MPNST) is a rare and aggressive soft tissue sarcoma for which effective treatments have not yet been established. The purpose of this study was to investigate a potential therapeutic role of COX-2 in MPNST.
Methods: We evaluated the expression of COX-2 in 44 cases of high-grade MPNST using immunohistochemical staining and compared the staining results with the characteristics and outcome of the patients. We also investigated the antitumor effect of etodolac, a selective COX-2 inhibitor, on MPNST cells in vitro using the MPNST cell line, FMS-1.
Results: Overexpression of COX-2 (>= 50% positive cells) was observed in 29 cases (65.9%), was significantly associated with a poor overall survival (P = 0.0495), and was considered an independent risk factor for a poor outcome by the results of both univariate and multivariate analysis. Etodolac induced apoptosis of FMS-1 cells through the activation of caspase-8, -9, and -3. Moreover, several caspase inhibitors significantly inhibited etodolac-induced apoptosis.
Conclusions: Selective COX-2 inhibitors including etodolac had an antitumor effect on MPNST cells, and their use holds promise as a novel therapeutic strategy for patients with MPNST to improve their prognoses.

DOI： 10.1371/journal.pone.0088035

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

Secondary rhabdomyosarcoma (RMS) after treatment of osteosarcoma (OS) is rare. Reported here is the case of a metachronous RMS in the nasal cavity, developing 12 years after successful treatment of non-metastatic OS. The patient was diagnosed as having OS of the femur at 2 years of age. Chemotherapy for OS included doxorubicin (cumulative dose, 488 mg/m(2)). No radiotherapy was given. There was no family history suggestive of cancer predisposition syndrome. At 14 years of age, alveolar RMS was diagnosed on histopathology. PAX3-FKHR fusion transcripts were detected on reverse transcription-polymerase chain reaction. Germline TP53 mutation was not seen on standard DNA sequencing. The occurrence of secondary sarcomas, in the Children's Cancer Survivor study conducted in North America, has been associated with high cumulative doses of anthracyclines, which may also have played a role in the development of RMS in the present case. In the future, novel molecular technologies might uncover genetic cancer predisposition in patients with metachronous cancers.

DOI： 10.1111/ped.12070

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記述言語：英語   掲載種別：研究論文（学術雑誌）  

DOI： 10.1186/1471-2407-13-224

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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  

The aim of the study was to determine the effect of alendronate on resorption of beta-tricalcium phosphate (beta-TCP) and bone formation in rats with adjuvant-induced arthritis (AIA). After preparation of a model of AIA in rats (day 0), alendronate or vehicle was injected intraperitoneally once daily five times in a week. Cylindrical beta-TCP was implanted into the rat femoral condyle on day 7. Rats were killed on days 12, 15, and 21, and specimens and serum samples were collected. Specimens were analyzed by tartrate-resistant acid phosphate (TRAP) staining, immunohistochemistry of the ED1 protein, and in situ hybridization with digoxigenin-labeled alpha 1 chain of type I procollagen (COL1A1). Mineralized bone sections were analyzed by Villanueva bone stain. The serum osteocalcin level was measured using an enzyme-linked immunosorbent assay kit. Alendronate decreased the number of TRAP-positive cells attached to beta-TCP, the numbers of ED1-positive multinucleated giant cells, and resorption of beta-TCP. In AIA rats treated with alendronate, COL1A1 mRNA-positive cells adhered to beta-TCP were round or cuboid whereas the cells in untreated AIA rats were fibroblast-like cells. Alendronate increased calcification of newly formed bone whereas it did not restore the bone formation suppressed with inflammation. These results suggest that alendronate has the potential to conduct mature bone after implantation of beta-TCP in AIA. Alendronate may help to reduce insufficiency of newly formed bone after implantation of beta-TCP in diseases characterized by increased bone resorption such as rheumatoid arthritis.

DOI： 10.1007/s00774-012-0369-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：W B SAUNDERS CO-ELSEVIER INC  

A case is presented of extensive alveolar bone grafting in a patient with bilateral cleft lip and palate and polyostotic fibrous dysplasia. The patient previously underwent bisphosphonate therapy. Because of an abnormal and often decreased bone turnover caused by the fibrous dysplasia and the bisphosphonate therapy, bone grafting in such a patient poses several potential difficulties. In addition, the histomorphometric analysis of the bone grafts showed markedly decreased bone turnover. However, alveolar bone grafting using the iliac crest was performed successfully. Sufficient occlusion was achieved by postoperative low-loading orthodontic treatment. (C) 2012 American Association of Oral and Maxillofacial Surgeons J Oral Maxillofac Surg 70:e500-e508, 2012

DOI： 10.1016/j.joms.2012.05.015

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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  

In the present study, we evaluated the safety and effectiveness of SYT-SSX-derived peptide vaccines in patients with advanced synovial sarcoma. A 9-mer peptide spanning the SYT-SSX fusion region (B peptide) and its HLA-A*2402 anchor substitute (K9I) were synthesized. In Protocols A1 and A2, vaccines with peptide alone were administered subcutaneously six times at 14-day intervals. The B peptide was used in Protocol A1, whereas the K9I peptide was used in Protocol A2. In Protocols B1 and B2, the peptide was mixed with incomplete Freund's adjuvant and then administered subcutaneously six times at 14-day intervals. In addition, interferon-a was injected subcutaneously on the same day and again 3 days after the vaccination. The B peptide and K9I peptide were used in Protocols B1 and B2, respectively. In total, 21 patients (12 men, nine women; mean age 43.6 years) were enrolled in the present study. Each patient had multiple metastatic lesions of the lung. Thirteen patients completed the six-injection vaccination schedule. One patient developed intracerebral hemorrhage after the second vaccination. Delayed-type hypersensitivity skin tests were negative in all patients. Nine patients showed a greater than twofold increase in the frequency of CTLs in tetramer analysis. Recognized disease progression occurred in all but one of the nine patients in Protocols A1 and A2. In contrast, half the 12 patients had stable disease during the vaccination period in Protocols B1 and B2. Of note, one patient showed transient shrinkage of a metastatic lesion. The response of the patients to the B protocols is encouraging and warrants further investigation.

DOI： 10.1111/j.1349-7006.2012.02370.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：ELSEVIER SCIENCE INC  

In ongoing clinical research into the use of cultured autogenous periosteal cells (CAPCs) in alveolar bone regeneration, CAPCs were grafted into 33 sites (15 for alveolar ridge augmentation and 18 for maxillary sinus lift) in 25 cases. CAPCs were cultured for 6 weeks, mixed with particulate autogenous bone and platelet-rich plasma, and then grafted into the sites. Clinical outcomes were determined from high-resolution three-dimensional computed tomography (3D-CT) images and histological findings. No serious adverse events were attributable to the use of grafted CAPCs. Bone regeneration was satisfactory even in cases of advanced atrophy of the alveolar process. Bone biopsy after bone grafting with CAPCs revealed prominent recruitment of osteoblasts and osteoclasts accompanied by angiogenesis around the regenerated bone. 3D-CT imaging suggested that remodeling of the grafted autogenous cortical bone particles was faster in bone grafting with CAPCs than in conventional bone grafting. The use of CAPCs offers cell-based bone regeneration therapy, affording complex bone regeneration across a wide area, and thus expanding the indications for dental implants. Also, it enables the content of particulate autogenous bone in the graft material to be reduced to as low as 40%, making the procedure less invasive, or enabling larger amounts of graft materials to be prepared. It may also be possible to dispense with the use of autogenous bone altogether in the future. The results suggest that CAPC grafting induces bone remodeling, thereby enhancing osseointegration and consequently reducing postoperative waiting time after dental implant placement. (C) 2012 Elsevier Inc. All rights reserved.

DOI： 10.1016/j.bone.2012.02.631

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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  

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

Objective: Dedifferentiated liposarcomas usually occur in the retroperitoneal space and relatively rarely in the extremities.
Methods: We identified 18 patients with primary dedifferentiated liposarcoma in the extremities from the files of Tohoku Musculoskeletal Tumor Society and analyzed demographics, histologic findings, treatments and prognostic factors. The average follow-up period was 58 months.
Results: The subjects were 12 men and 6 women with a mean age of 65 years. All tumors were in the thigh. Nine patients noticed a rapid enlargement of the long-standing tumor. Histologic subtypes of the dedifferentiated area were undifferentiated pleomorphic sarcoma (n = 12), osteosarcoma (n = 2), rhabdomyosarcoma (n = 2), leiomyosarcoma (n = 1) and malignant peripheral nerve sheath tumor (n = 1). In the patient with rhabdomyosarcoma-like dedifferentiated area, extensive necrosis was observed after the preoperative chemotherapy. One patient who underwent marginal excision developed a local recurrence, but inadequate surgical margin was not associated with a risk of local recurrence. Three patients had lung metastasis at initial presentation, and four other patients developed lung metastases during the follow-up period. The overall survival rate was 61.1% at 5 years. On univariate analyses, large size of the dedifferentiated area (>8 cm), high MIB-1-labeling index (>30%) for the dedifferentiated area and lung metastasis at initial presentation were significantly associated with poor prognosis.
Conclusions: Primary dedifferentiated liposarcoma in the extremities predominantly occurred in the thigh and a rapid enlargement of long-standing tumors was a characteristic symptom. Although the local behavior of these tumors was less aggressive than that of retroperitoneal dedifferentiated liposarcomas, they had a relatively high metastatic potential.

DOI： 10.1093/jjco/hyr098

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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：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：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：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：MOSBY-ELSEVIER  

DOI： 10.1016/j.jse.2010.05.017

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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  

Poorly differentiated type synovial sarcoma (PDSS) is a variant of synovial sarcoma characterized by predominantly round or short-spindled cell morphology. Although accumulating evidence from clinicopathologic studies suggests a strong association between this variant of synovial sarcoma and poor prognosis, little has been reported on the molecular basis of PDSS. To gain insights into the mechanism(s) that underlie the emergence of PDSS, we analyzed the gene expression profiles of 34 synovial sarcoma clinical samples, including 5 cases of PDSS, using an oligonucleotide microarray. In an unsupervised analysis, the 34 samples fell into 3 groups that correlate closely with histologic subtypes: monophasic, biphasic, and poorly differentiated types. PDSS was characterized by down-regulation of genes associated with neuronal and skeletal development and cell adhesion. Moreover, upregulation of genes on a specific chromosomal locus, 8q21.11, was identified. This locus-specific transcriptional activation in PDSS was confirmed by reverse transcriptase-PCR analysis of 9 additional synovial sarcoma samples. Our results indicate that PDSS tumors constitute a distinct group based on expression profiles.

DOI： 10.1097/PAS.0b013e3181f7ce2c

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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  

Imatinib myselate is a molecularly targeted drug that inhibits Abl tyrosine kinase, as well as type III tyrosine kinase receptors such as platelet-derived growth factor receptor (PDGFR), KIT, colony-stimulating factor 1 receptor (CSF-1R), and discoidin domain receptor (DDR). Ph1 chromosome-positive chronic myeloid leukemias (CMLs), KIT-positive gastrointestinal stromal tumors (GISTs), and PDGFR-positive dermatofibrosarcoma protuberans (DFSP) have been reported to be responsive to imatinib treatment. We conducted a multicenter Phase II trial of imatinib in patients with relapsed or refractory KIT-positive (excluding GISTs) or PDGFR-positive sarcomas.
Patient ages ranged from 12 and 75 years. Eligibility criteria included (1) metastatic sarcomas with a definitive diagnosis based on histopathology or that were completely unresectable and locally advanced; (2) relapsed or refractory cases that had completed standard treatment; and (3) a tumor confirmed by immunohistochemical staining to be KIT- or PDGFR-positive. A 600-mg dose of imatinib was administered to patients once a day, with each patient receiving six courses of the drug and each course lasting 4 weeks. In cases categorized as stable or progressive, the imatinib dose was increased to 800 mg/day administered twice daily.
A total of 25 patients who met the eligibility criteria were enrolled in the trial; 22 were evaluated for response. The response rate with a 600 mg/day dose of imatinib was 4.5% (0 complete response, 1 partial response). There were no other objective responses after increasing imatinib to 800 mg/day (0/10). We estimated 50% progression-free survival to be 61.0 days for an imatinib dose of 600 mg/day based on the Kaplan-Meier method. Side effects of imatinib were generally similar to those observed in previous clinical trials.
Our results did not indicate effectiveness of imatinib monotherapy at a dose of 600 or 800 mg/day in patients with relapsed or refractory KIT-positive (excluding GISTs) or PDGFR-positive sarcomas. Our findings suggest the need to evaluate the synergistic effect of combination therapy with other anticancer drugs.

DOI： 10.1007/s00776-010-1506-9

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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：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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DOI： 10.1186/1756-9966-28-44

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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：TAYLOR & FRANCIS INC  

Strategies effective for accelerating methotrexate removal in delayed methotrexate excretion have not been universally accepted. The authors report a case of a 12-year-old girl with osteosarcoma who developed acute renal failure immediately after the first administration of high-dose methotrexate. Plasma methotrexate was effectively removed with repeated charcoal hemoperfusion in addition to plasma exchange and leucovorin rescue. Charcoal hemoperfusion was most effective for reducing plasma methotrexate with approximately 50% of methotrexate being reduced during each of the procedures. No rebound increase in MTX levels was observed. The patient received further therapy with other cancer drugs and has been well for 3.5 years.

DOI： 10.1080/08880010902976023

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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: Despite accumulating knowledge of chimeric genes derived from fusion of the HMGA2 gene with multiple partners in lipomas, the different clinicopathological features of lipomas depending on different gene aberrations have not been well documented. The purpose of this study was to examine the clinical significance of the expression of fusion genes in lipomas. Patients and Methods: The expressions of three previously reported gene fusion transcripts, including HMGA2/LPP, HMGA2/RDC1 and HMGA2/NFIB, were analyzed in 102 tumors from patients with lipomas. Results: There were 23 cases (22.5%) expressing HMGA2/LPP, 2 cases (1.9%) expressing HMGA2/RDC1 and no cases of HMGA2/NFIB expression (0%). There were no significant intergroup differences in age, gender, body mass index, tumor size or location. The magnetic resonance images and pathological features were also not different in regard to the status of fusion gene expression. Conclusion: There were no significant differences of clinicopathological features inpatients with lipoma with or without these fusion gene transcripts.

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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  

Background. Lipoma-like liposarcomas mimic deep-seated lipomas in regard to imaging as well as histological findings and occasionally cause problems concerning diagnosis and treatment. The differences in the imaging findings among these lesions are not well defined. The purpose of this study was to elucidate the differences among the deep-seated adipocytic neoplasms including intramuscular lipoma, intermuscular lipoma, and lipoma-like liposarcoma.
Methods. The imaging and clinicopathological findings of 40 intramuscular lipomas, 27 intermuscular lipomas, and 22 lipoma-like liposarcomas were evaluated, and the possibilities in the differential diagnosis were assessed.
Results. Although the most frequent symptom was a palpable mass, swelling was a common symptom of intramuscular lipomas and lipoma-like liposarcomas. Imaging studies revealed dumbbell-shaped appearances among intermuscular lipomas, whereas spherical masses were characteristic of intramuscular lipomas and lipoma-like liposarcomas. Computed tomography and magnetic resonance imaging revealed fatty lesions containing streaky structures in benign lesions, and CT revealed foci of hazy amorphous density, representing spindle cell proliferation, in lipoma-like liposarcoma. Although streaky structures corresponding to entrapped muscle fibers were thick and occasionally interrupted in intramuscular lipomas, the streaky structures corresponding to areolar fibrous tissue were thin and were usually not interrupted in intermuscular lipomas. In lipoma-like well-differentiated liposarcomas, thick streaks represented entrapped muscle fibers, and thin streaks represented fibrous tissue or neoplastic spindle cell proliferation.
Conclusions. The imaging findings are helpful and often afford almost pathognomonic evidence of these lesions and could help with the selection of appropriate surgery.

DOI： 10.1007/s00776-007-1177-3

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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：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：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/s00776-007-1125-2

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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：LIPPINCOTT WILLIAMS & WILKINS  

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

DOI： 10.1097/01.brs.0000251045.79708.7a

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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：TAYLOR & FRANCIS AS  

Osteosarcoma is the most common form of malignant bone tumor that occurs during childhood and adolescence. The proximal fibula is a relatively rare site for osteosarcoma. We reviewed 305 cases of osteosarcoma registered at the Tohoku Musculoskeletal Tumor Society (TMTS) between 1975 and 1999. Thirteen patients (4.3%) had their osteosarcomas localized in the proximal fibula. Conventional fibroblastic osteosarcoma accounted for 46% of the cases in this series. Limb-sparing surgery was performed in all 13 patients during initial surgery, and the peroneal nerve was preserved in 4 cases. These 4 cases developed local recurrences, but additional wide excision or radiation had a beneficial effect on the recurrences. In our series, the patients showed a 5-year survival rate 76 per cent. The postoperative function of the knee remained good despite various reattachment procedures of lateral co-lateral ligament. As well as resection of the proximal fibula, our results indicate that osteosarcoma of the proximal fibula has a good prognosis for cases who undergo adequate initial surgery.

DOI： 10.3109/2000-1967-209

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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  

We analyzed the effect of glucocorticoid on bone regeneration after bone marrow ablation in tibiae of 8-week-old rats. Methylprednisolone sodium succinate (MPSS) was injected intramuscularly at a dose of 100 mg/kg/day for 3 days. Tibiae on days 1, 3, 5, 7, 10, 12, and 14 after ablation were subjected to tartrate-resistant acid phosphatase staining, immunohistochemistry, in situ hybridization, and transmission electron microscopy (TEM), and measurement of the volume of newly-formed bone and the osteoclast number. MPSS significantly decreased the newly-formed bone volume on day 7, and immature bone still remained on day 10 in the MPSS-treated group. The volume of this bone was significantly higher than that in the control group. However, there were no differences between the groups in the osteoclast number, the expression of mRNAs for osteoblast differentiation markers, and alkaline phosphatase and cathepsin K judged by immunohistochemistry. TEM findings showed no difference in the form of osteoblasts, whereas osteoclasts in the MPSS-treated group had less developed ruffled borders, compared to those in the control group. These results suggest that MPSS treatment affects neither the differentiation nor the shape of osteoblasts, and does not change the osteoclast number or the cathepsin K level. However, high dose MPSS inhibits both bone formation and resorption during bone regeneration after rat tibial bone marrow ablation, and inhibits ruffled border formation in osteoclasts. These data will be useful to develop bone regenerative therapies for bone diseases due to high dose steroid administration.

DOI： 10.1002/jemt.20355

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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  

The aim of the study was to examine the chronological histology of osteoinduction of highly purified beta-tricalcium phosphate (beta-TCP) implanted in dog dorsal muscles. Specimens were harvested on days 14, 28, 42, 56, 112 and 168 after implantation, and were analyzed by hernatoxylin and eosin (HE) staining, tartrate-resistant acid phosphatase (TRAP) staining, immunohistochemistry, in situ hybridization, and silver impregnation. After day 28, abundant TRAP- and cathepsin K-positive multinucleated cells adhered to beta-TCP, suggesting that these cells are osteoclasts that can resorb beta-TCP. On day 56, new bone was formed and alpha 1 chain of type I procollagen mRNA-positive osteoblasts lined the newly formed bone. Silver impregnation showed abundant collagen fibrits within the beta-TCP micropores. These results suggest that micropores function as a storage space for extracellular matrix components, including collagen. Newly formed bone never degenerated in the late stage, suggesting that beta-TCP has good biocompatibility and this material retains the conditions appropriate for osteointegration and bioresorption. In conclusion, beta-TCP has osteoinductivity after implantation in dog dorsal muscles without use of bone marrow cells or osteoinductive cytokines. The appearance of a large number of active osteoclasts precedes new bone formation. (c) 2006 Elsevier Ltd. All rights reserved.

DOI： 10.1016/j.biomaterials.2006.04.016

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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：ELSEVIER SCI LTD  

The aim of this study was to examine the chronological histology associated with highly purified beta-tricalcium phosphate (beta-TCP) implanted in the rat Femoral condyle. Specimens were harvested on days 4, 7, 14, 28 and 56 after implantation, and were analyzed by tartrate-resistant acid phosphatase (TRAP) staining, immunohistochemistry of the EDI protein as a marker of the phagocyte system,and in situ hybridization with digoxigenin-labeled alpha 1 chain of type 1 procollagen (COL1A1),osteopontinand osteocalcin.beta-TCP was resorbed in a chronological manner. Although new bone was not observed on day 4, fibroblast-like cells around beta-TCP were positive for COL1A1 and osteopontin mRNA. New bone formation presented after day 7. In the double-staining for OPN and EDI on day 7, most cells around beta-TCP were positive for either osteopontin mRNA or ED1 protein. However, there were some doubly positive multinucleated cells, suggesting that they belonged to the mononuclear phagocyte system. After day 28, the implanted region was replaced with bone marrow. Multinucleated TRAP-positive and ED1-positive cells which adhered to beta-TCP at all stages seemed to be osteoclasts and they continuously resorbed beta-TCP. beta-TCP has a good biocompatibility since both bioresorption and bone formation started at an early stage after implantation. (c) 2005 Elsevier Ltd. All rights reserved.

DOI： 10.1016/j.biomaterials.2005.02.026

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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：ELSEVIER SCIENCE INC  

Clear cell chondrosarcoma is a rare cartilaginous tumor of low-grade malignancy that most often arises in the epiphysis of long bones in the third and fourth decades of life. Cytogenetic studies of clear cell chondrosarcoma are few. In this study, the cytogenetic findings of 4 cases of clear cell chondrosarcoma are presented. Clonal chromosomal abnormalities were detected in 3 cases. A tumor specific anomaly was not identified, however, extra copies of chromosome 20 and loss or rearrangements of 9p appear to be recurrent. (c) 2005 Elsevier Inc. All rights reserved.

DOI： 10.1016/j.cancergencyto.2005.03.003

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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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DOI： 10.1200/JCO.2005.05.042

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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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DOI： 10.1158/1078-0432.CCR-04-2375

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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：TAYLOR & FRANCIS AS  

Infantile fibrosarcoma is a rare soft tissue malignant tumor, when it occurs, it is usually seen in the first year of life. The clinical course of infantile fibrosarcoma is more favorable and metastasis is rare compared with that in adulthood. While adult fibrosarcoma are common in the thigh, infantile fibrosarcoma affect chiefly the distal portions of the extremities. Standard treatment is primarily wide surgical excision. In this case report, we present our experience of an infantile fibrosarcoma of thigh with good clinical course 36 months after tumor resection and the usefulness of detecting the ETV6-NTRK3 gene fusion in differential diagnosis.

DOI： 10.3109/2000-1967-184

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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  

Background: The clinicopathologic profile and prognostic factors of osteosarcomas after the age of 50 years have been obscure.
Methods: Clinicopathologic features were analyzed in 645 patients with osteosarcoma who were registered at the Tohoku Musculoskeletal Tumor Society and National Cancer Center in Tokyo between 1972 and 2002.
Results: Sixty-four patients (9.9%; 34 men and 30 women) were more than 50 years old. The most common location was the distal femur (n = 13), followed by the pelvis (n = 10), proximal femur (n = 9), and proximal fibula (n = 6). Seven (11%) patients had lung metastasis at initial presentation. On radiographs, an osteolytic appearance without periosteal reactions was a common and characteristic feature. Forty-eight tumors (75%) were classified as conventional osteosarcomas. Fourteen cases (22%) were secondary; postradiation osteosarcoma was most common in these patients, but there was no Paget's sarcoma. At the initial presentation, misdiagnoses without biopsy were made in 15 (23%) of the 64 cases. Preoperative chemotherapy was given to 22 patients, but the effect was poor in 18 cases (82%). Fifty-four patients underwent surgery, whereas the other 10 patients were treated without surgery because of systemic or inoperable local conditions. The overall survival rate at 5 years was 55.5%. Multivariate. analysis showed initial pulmonary metastasis, axial tumor location, and larger tumors as significant prognostic factors.
Conclusions: In northern Japan, most patients with osteosarcoma after the age of 50 had primary osteosarcoma. Careful radiological examination and biopsy are mandatory for correct diagnosis. Current systemic chemotherapy is not effective for this age group. Alternative treatment strategies should be considered.

DOI： 10.1245/ASO.2004.03.004

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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：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：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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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：LIPPINCOTT WILLIAMS & WILKINS  

Objective: To describe clinical and imaging findings of distended scapulothoracic bursitis without scapular snapping, which is often confused with a soft tissue tumor.
Methods: Nine patients (6 male, 3 female; age range: 50-73 years; mean age = 67 years) with distended scapulothoracic bursitis diagnosed by clinical and magnetic resonance (MR) imaging findings were studied. The results of a histologic examination were available in 1 case.
Results: All patients presented with painless palpable masses below the scapula, and the initial diagnoses were soft tissue tumors. On MR images, the lesions were 5.5 to 12 cm in maximum diameter (mean = 7.7 cm) and well-demarcated cystic masses situated in the subscapular region between the serratus anterior and the chest wall. There was no solid portion on the cyst walls. The findings of hemorrhage within the bursae were present in all cases. Every mass regressed in size spontaneously after a few to several weeks, and no lesions revealed any malignant findings of sarcomas.
Conclusions: Distended scapulothoracic bursae without scapular snapping resemble soft tissue tumors. They have some specific MR findings; therefore, precise recognition of these findings is important to avoid misdiagnoses and unnecessary treatments.

DOI： 10.1097/00004728-200403000-00012

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

DOI： 10.1093/jjco/hyh027

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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  

The objective of this study was to review the clinicopathological features of seven patients presenting with well-differentiated liposarcoma (WDL), which was associated with subcutaneous lipoma. From 1980 through 2002, 34 individuals displaying WDL were treated in our institutions. Lipoma was observed in seven of these 34 patients (five men and two women, mean age of 66.7 years). The rate of coexistence of lipoma in WDL [20.6% (7/34)] cases was significantly higher than the corresponding rate in the other liposarcoma subtypes [2.5% (1/40)]. Immunohistochemically, cdk4 was positive in all WDLs (100%). ki-67 was positive in 571% (4/7) and mdm2 and p53 were positive in 14.5% (1/7) of the WDL cases. Weak cdk4 immunoreactivity was detected in two lipomas. All lipomas were negative for mdm2, p53 and ki-67. Comparison of the expression profile in these malignant and benign tumors, which had arisen in identical genetic backgrounds, confirmed the involvement of these proteins, especially cdk4, in the tumorigenesis process of WDL.

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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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DOI： 10.1097/01.BRS.0000092344.14376.D9

PubMed

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記述言語：英語   出版者・発行元：SPRINGER-VERLAG  

DOI： 10.1007/s00428-003-0789-z

Web of Science

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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：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：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

Scopus

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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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DOI： 10.2106/00004623-200203000-00015

PubMed

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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  

We report six patients with soft tissue sarcoma mimicking traumatic hematoma. The lesions in these patients, showed huge hematomas and were characterized by rapid growth. Cytology of percutaneous
aspiration biopsy samples was performed in all six patients
however, in five patients, findings for malignant cells were negative. Consequently, they were misdiagnosed, resulting in a poor prognosis. We conducted a retrospective study in which we evaluated the clinical findings, the magnetic resonance (MR) images, and computed tomography (CT) scans of the soft tissue sarcomas forming huge hematomas in the lesion. MR imaging revealed the fine tumor mass with enhancement and characterized the hematoma in the lesion in a more precise fashion than did CT. We conclude that MR imaging is a suitable method for differentiating these soft tissue sarcomas from chronic traumatic hematoma.

DOI： 10.1007/s776-002-8410-5

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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  

当院で2015年6月〜2016年5月に大腿骨近位部骨折手術を行った200例(骨折群)と、骨粗鬆症に対し外来治療中で大腿骨近位部骨折の既往がない患者のうち骨折群と年齢を対応させた200例(対照群)について内服薬使用状況を調査し、群間比較した。結果、1例あたりの平均内服薬数は骨折群5.5個、対照群5.0個であり、骨折群のほうが多い傾向にあった。症例ごとの内服薬数別に、0個、1〜2個、3〜4個、5〜6個、7〜8個、9個以上に区分すると、骨折群は7個以上の症例割合が高い傾向にあった。転倒を誘発しうるとされている薬剤別にみると、骨折群は「抗精神病薬」「利尿薬」「抗てんかん薬」「血管拡張薬」「睡眠薬」を内服している割合が有意に高かった。

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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：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：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：(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：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)日本内分泌学会  

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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：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：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：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：(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：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  

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：(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：(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：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：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：(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：(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：(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  

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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  

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：(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)日本皮膚科学会  

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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：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：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：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：日本語   出版者・発行元：新潟県医師会  

癌を扱う医師であれば避けて通れないのが骨転移であり、癌検診に必ずしも精通していない整形外科医にとっても骨転移に迅速かつ柔軟に対応することが求められている。最近では、骨転移に対する薬物治療にも注目が集まっている。骨粗鬆症に使用される薬剤と、投与量は違うが本質は同じものである。骨転移の疫学、診断、薬物治療、放射線治療、手術治療、について述べた。

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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  

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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：(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：(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  

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：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：(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：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：日本語   出版者・発行元：新潟整形外科研究会  

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DOI： 10.1302/0301-620X.80B4.8532

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DOI： 10.1002/(SICI)1097-0142(19970701)80:1<50::AID-CNCR7>3.3.CO;2-R

PubMed

CiNii Article

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