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DOI： 10.1186/s13047-022-00582-z

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DOI： 10.7554/eLife.73539.

Language：English   Publishing type：Research paper (scientific journal)  

DOI： 10.1186/s13075-022-02746-5

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Language：English   Publishing type：Research paper (scientific journal)  

DOI： 10.1016/j.injury.2021.07.039.

Language：English   Publishing type：Research paper (scientific journal)  

DOI： 10.1186/s41927-020-00164-1.

Language：English   Publishing type：Research paper (scientific journal)  

DOI： 10.3390/jcm9103133

Language：Japanese   Publishing type：Research paper (scientific journal)  

Language：English   Publishing type：Research paper (scientific journal)  

Language：English   Publisher：Skeletal Radiology  

Synovial chondrosarcoma (CHS) is a rare malignant tumor arising from the synovial tissue and is often associated with synovial chondromatosis. Herein, we present a unique case of primary synovial CHS in the shoulder joint without evidence of synovial chondromatosis. A 60-year-old man presented to our hospital with a complain of left shoulder pain that persisted for 6 years, which was initially misdiagnosed as synovitis. Radiography revealed an osteolytic lesion involving the humerus and the scapula. Histologically, the tumor exhibited features of grade 2 synovial CHS, infiltrating the trabecular bones and intra-articular space. Wide resection led to a 9-year recurrence-free survival. This case underscores the challenges in diagnosing and managing synovial CHS, particularly in cases with atypical presentations lacking synovial chondromatosis, necessitating careful follow-up and adequate surgical intervention.

DOI： 10.1007/s00256-024-04811-3

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Language：English  

DOI： 10.1016/j.esmoop.2024.103726

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Language：English   Publisher：Scientific Reports  

The coronavirus disease (COVID-19) pandemic negatively affected the diagnosis and treatment of several cancer types. However, this pandemic’s exact impact and extent on bone and soft tissue sarcomas need to be clarified. We aimed to investigate the effect of the COVID-19 pandemic and emergency declaration by the local government on consultation behavior and clinical stage at diagnosis of bone and soft tissue sarcoma. A total of 403 patients diagnosed with bone and soft tissue sarcoma who initially visited three sarcoma treatment hospitals between January 2018 and December 2021 were included. The monthly number of newly diagnosed soft tissue sarcoma patients was reduced by 25%, and the proportion of soft tissue patients with stage IV disease at diagnosis significantly increased by 9% during the COVID-19 pandemic compared to before the COVID-19 pandemic. Furthermore, the monthly number of new primary bone and soft tissue sarcoma patients significantly decreased by 43% during the state of emergency declaration. The COVID-19 pandemic had a negative impact on soft tissue sarcoma patients’ consultation behavior and increased the proportion of advanced-stage patients at initial diagnosis. An emergency declaration by the local government also negatively affected primary bone and soft tissue sarcoma patients’ consultation behavior.

DOI： 10.1038/s41598-024-71830-4

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Language：English  

DOI： 10.1093/jjco/hyae095

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Language：English  

DOI： 10.7759/cureus.64458

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Language：English   Publisher：(公社)日本整形外科学会  

Language：English  

DOI： 10.1093/mr/roae048

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Language：English   Publisher：Taylor & Francis  

Publisher：南江堂  

DOI： 10.15106/j_seikei75_487

CiNii Research

Language：English   Publisher：(公社)日本整形外科学会  

Publisher：金原出版  

DOI： 10.18888/se.0000002976

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Language：English   Publisher：British Journal of Cancer  

Background: Leiomyosarcomas are among the most common histological types of soft tissue sarcoma (STS), with no effective treatment available for advanced patients. Lung metastasis, the most common site of distant metastasis, is the primary prognostic factor. We analysed the immune environment targeting lung metastasis of STS to explore new targets for immunotherapy. Methods: We analysed the immune environment of primary and lung metastases in 38 patients with STS using immunohistochemistry. Next, we performed gene expression analyses on primary and lung metastatic tissues from six patients with leiomyosarcoma. Using human leiomyosarcoma cell lines, the effects of the identified genes on immune cells were assessed in vitro. Results: Immunohistochemistry showed a significant decrease in CD8+ cells in the lung metastases of leiomyosarcoma. Among the genes upregulated in lung metastases, epithelial cellular adhesion molecule (EPCAM) showed the strongest negative correlation with the number of CD8+ cells. Transwell assay results showed that the migration of CD8+ T cells was significantly increased in the conditioned media obtained after inhibition or knock down of EPCAM. Conclusions: EPCAM was upregulated in lung metastases of leiomyosarcoma, suggesting inhibition of CD8+ T cell migration. Our findings suggest that EPCAM could serve as a potential novel therapeutic target for leiomyosarcoma.

DOI： 10.1038/s41416-024-02576-z

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Language：English   Publisher：Immunological Medicine  

Synovial tissue-resident macrophages (STRMs) maintain normal joint homeostasis in a steady state. However, it is unclear whether STRMs still play homeostatic roles or change the functions in the joint of rheumatoid arthritis (RA), where infiltrating peripheral blood monocyte-derived macrophages (PBMoMs) play proinflammatory roles. In the present study, we examined changes in the phenotypes and functions of STRMs in response to RA-related stimuli in vitro. STRMs were prepared from non-inflammatory osteoarthritis (OA) joint synovium, which is histologically indistinguishable from normal joint synovium. PBMoMs were prepared and used for comparison. After stimulation with plate-bound IgG, which mimics anti-citrullinated protein antibody immunocomplex formed in RA joints, or with combinations of RA-related inflammatory mediators, namely tumor necrosis factor-α (TNF-α) and prostaglandin E2 or interferon-γ, PBMoMs downregulated surface markers and genes associated with anti-inflammatory macrophages, and upregulated cytokine and marker genes of proinflammatory macrophages in RA. On the other hand, STRMs hardly changed the expression of surface molecules and marker genes but altered the pattern of cytokine gene expression after stimulation like PBMoMs. Furthermore, in vitro stimulated STRMs promote proinflammatory functions of cocultured synovial fibroblasts. Thus, STRMs might play proinflammatory roles in RA joints, while maintaining their phenotypes in the steady state.

DOI： 10.1080/25785826.2023.2300853

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Language：Japanese   Publisher：西日本整形・災害外科学会  

<p>2020年の関節リウマチ診療ガイドラインでは，人工足関節全置換術（total ankle arthroplasty，TAA）は足関節固定と同等レベルの推奨とされているが，実臨床においては足関節固定術が第一選択とされることが多い．当院ではRA足関節障害に対し外側侵入型TAA（TM ankle）を用いた治療を行なっており，今回この使用経験について報告する．2019～2022年に関連施設でTAAを行ったRA患者6例6足を対象に，足関節可動域，術後合併症，JSSFスコアについて検討した．術後最終経過観察時のJSSFは術前より有意に改善していた．可動域の合計は平均29.2°であった．合併症は2名で外果プレートに感染を生じたが，人工関節そのものに感染は至らず，またインプラントのゆるみは発生していない．RA足関節障害に対し外側進入型TAAを施行し，短期ではあるが良好な成績を得ており，今後足関節固定術に変わる手術法として期待できると考える．</p>

DOI： 10.5035/nishiseisai.73.170

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Language：English   Publisher：Modern Rheumatology  

Objective: To compare the effectiveness of methotrexate (MTX) as initial therapy in patients with late-onset and younger-onset rheumatoid arthritis (LORA and YORA). Methods: Of 114 patients with YORA and 96 patients with LORA, defined as RA occurring at ≥65 years of age, enrolled in a multicentre RA inception cohort study, 71 and 66 patients who had been followed up to 6 months after starting MTX treatment were included in this study. Results: Proportions of patients on MTX treatment at 6 months were 96% and 92% in the YORA and LORA groups, respectively. Despite lower doses of MTX in the LORA group compared with the YORA group, no significant difference was observed in clinical disease activity index scores between the two groups throughout the follow-up period. The proportion of patients in clinical disease activity index remission at 6 months was 35% in both groups. Logistic regression analysis revealed that knee joint involvement and high Health Assessment Questionnaire-Disability Index were significant negative predictors of achieving clinical disease activity index remission at 6 months in the LORA group. Conclusion: Observations up to 6 months revealed that the effectiveness of MTX administered based on rheumatologist discretion in patients with LORA is comparable to that in patients with YORA in clinical settings.

DOI： 10.1093/mr/roae027

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Language：English   Publisher：Oxford University Press  

Language：Japanese   Publisher：西日本整形・災害外科学会  

2020年の関節リウマチ診療ガイドラインでは,人工足関節全置換術(total ankle arthroplasty,TAA)は足関節固定と同等レベルの推奨とされているが,実臨床においては足関節固定術が第一選択とされることが多い.当院ではRA足関節障害に対し外側侵入型TAA(TM ankle)を用いた治療を行なっており,今回この使用経験について報告する.2019～2022年に関連施設でTAAを行ったRA患者6例6足を対象に,足関節可動域,術後合併症,JSSFスコアについて検討した.術後最終経過観察時のJSSFは術前より有意に改善していた.可動域の合計は平均29.2°であった.合併症は2名で外果プレートに感染を生じたが,人工関節そのものに感染は至らず,またインプラントのゆるみは発生していない.RA足関節障害に対し外側進入型TAAを施行し,短期ではあるが良好な成績を得ており,今後足関節固定術に変わる手術法として期待できると考える.(著者抄録)

Publisher：協和企画  

DOI： 10.24733/pd.0000003734

CiNii Research

Language：Japanese   Publisher：(株)協和企画  

＜文献概要＞症例のポイント　・体幹に発生し巨大化した皮下腫瘍であり,類表皮嚢腫や粘液癌,脂肪肉腫との鑑別を要した.・全切除標本の病理組織学的診断で診断確定に至った.

Language：English   Publisher：npj Precision Oncology  

Prognosis after neoadjuvant chemotherapy (NAC) for osteosarcoma is generally predicted using manual necrosis-rate assessments; however, necrosis rates obtained in these assessments are not reproducible and do not adequately reflect individual cell responses. We aimed to investigate whether viable tumor cell density assessed using a deep-learning model (DLM) reflects the prognosis of osteosarcoma. Seventy-one patients were included in this study. Initially, the DLM was trained to detect viable tumor cells, following which it calculated their density. Patients were stratified into high and low-viable tumor cell density groups based on DLM measurements, and survival analysis was performed to evaluate disease-specific survival and metastasis-free survival (DSS and MFS). The high viable tumor cell density group exhibited worse DSS (p = 0.023) and MFS (p = 0.033). DLM-evaluated viable density showed correct stratification of prognosis groups. Therefore, this evaluation method may enable precise stratification of the prognosis in osteosarcoma patients treated with NAC.

DOI： 10.1038/s41698-024-00515-y

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DOI： 10.35848/1347-4065/acfd35

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Language：English   Publishing type：Research paper (scientific journal)  

Language：English   Publishing type：Research paper (scientific journal)  

DOI： 10.18494/SAM4767

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Language：English   Publisher：Cell Death Discovery  

In osteoclastogenesis, the metabolism of metal ions plays an essential role in controlling reactive oxygen species (ROS) production, mitochondrial biogenesis, and survival, and differentiation. However, the mechanism regulating metal ions during osteoclast differentiation remains unclear. The metal-binding protein metallothionein (MT) detoxifies heavy metals, maintains metal ion homeostasis, especially zinc, and manages cellular redox levels. We carried out tests using murine osteoclast precursors to examine the function of MT in osteoclastogenesis and evaluated their potential as targets for future osteoporosis treatments. MT genes were significantly upregulated upon differentiation from osteoclast precursors to mature osteoclasts in response to receptor activators of nuclear factor-κB (NF-κB) ligand (RANKL) stimulation, and MT3 expression was particularly pronounced in mature osteoclasts among MT genes. The knockdown of MT3 in osteoclast precursors demonstrated a remarkable inhibition of differentiation into mature osteoclasts. In preosteoclasts, MT3 knockdown suppressed the activity of mitogen-activated protein kinase (MAPK) and NF-κB signaling pathways upon RANKL stimulation, leading to affect cell survival through elevated cleaved Caspase 3 and poly (ADP-ribose) polymerase (PARP) levels. Additionally, ROS levels were decreased, and nuclear factor erythroid 2-related factor 2 (NRF2) (a suppressor of ROS) and the downstream antioxidant proteins, such as catalase (CAT) and heme oxygenase 1 (HO-1), were more highly expressed in the MT3 preosteoclast knockdowns. mitochondrial ROS, which is involved in mitochondrial biogenesis and the production of reactive oxygen species, were similarly decreased because cAMP response element-binding (CREB) and peroxisome proliferator-activated receptor γ coactivator 1β (PGC-1β) were less activated due to MT3 depletion. Thus, by modulating ROS through the NRF2 pathway, MT3 plays a crucial role in regulating osteoclast differentiation and survival, acting as a metabolic modulator of intracellular zinc ions. [Figure not available: see fulltext.]

DOI： 10.1038/s41420-023-01729-y

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Language：English   Publisher：Bone and Joint Open  

Aims To evaluate mid-to long-term patient-reported outcome measures (PROMs) of endoprosthetic reconstruction after resection of malignant tumours arising around the knee, and to investigate the risk factors for unfavourable PROMs. Methods The medical records of 75 patients who underwent surgery between 2000 and 2020 were retrospectively reviewed, and 44 patients who were alive and available for follow-up (at a mean of 9.7 years postoperatively) were included in the study. Leg length discrepancy was measured on whole-leg radiographs, and functional assessment was performed with PROMs (Toronto Extremity Salvage Score (TESS) and Comprehensive Outcome Measure for Musculoskeletal Oncology Lower Extremity (COMMON-LE)) with two different aspects. The thresholds for unfavourable PROMs were determined using anchor questions regarding satisfaction, and the risk factors for unfavourable PROMs were investigated. Results The thresholds for favourable TESS and COMMON were 64.8 and 70.4 points, respectively. Multivariate analysis showed that age at surgery (p = 0.004) and postoperative leg length discrepancy (p = 0.043) were significant risk factors for unfavourable TESS results, while age at surgery (p < 0.001) was a significant risk factor for unfavourable COMMON-LE results. Following receiver operating characteristic analysis, the threshold for both TESS and COMMON-LE was 29 years of age at surgery. Additionally, a leg length discrepancy of 8.2 mm was the threshold for unfavourable TESS. Conclusion Patients aged > 29 years at the time of surgery require appropriate preoperative counselling and adequate postoperative physical and socioemotional support. Reconstruction equivalent to the length of the resected bone can reduce the risk of functional disabilities in daily living.

DOI： 10.1302/2633-1462.412.BJO-2023-0125.R1

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Language：Japanese   Publishing type：Research paper (scientific journal)  

Language：Japanese   Publisher：(一社)日本足の外科学会  

リウマチ患者の前足部手術における創傷治癒遅延の現状を把握し,予測因手を明らかにするため,2010年5月から2021年12月の間に当院で前足部に対して手術を施行された79患者103足について後ろ向きに比較検討した.創傷治癒遅延の発生率は29.2%であった.DWHあり群では有意に高年齢,ESR1hr亢進,低Alb,低T-cholを認めた.DWHあり群ではMTXの利用割合が有意に低く,Larsen分類3以上の比率が高かった.手術因子に群間差を認めなかった.DWHあり群において,mGPS,CONUT scoreで有意に低い栄養状態を示した.多変量解析では,高齢,ESR1hr高値,CONUT score低値がDWH発生の危険因子であった.疾患活動性のコントロールおよび術前栄養状態の改善によって,RA前足部手術の創傷治癒遅延発生を減少させる可能性がある.(著者抄録)

Language：English   Publishing type：Research paper (scientific journal)  

DOI： 10.1186/s12885-023-10693-w

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Language：English   Publishing type：Research paper (scientific journal)  

DOI： 10.1016/j.jos.2023.03.020

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Language：Japanese   Publisher：西日本整形・災害外科学会  

<p>【目的】当院における関節リウマチ（RA）に対する人工膝関節全置換術（TKA）の患者背景の変遷を調査し，長期患者立脚評価を行った．【対象と方法】2001年～2015年に当院で施行されたTKA数の推移を調査した．同期間に施行されたRAに対するTKA（RA-TKA）158例で術前の患者背景を解析した．同158例から現在の年齢が80歳以下である52例72膝を抽出し，Knee Society Score（KSS）を送付した．返答のあった28例38膝でKSSの各項目（膝の症状，満足度，手術に関する期待度）における点数，各項目に影響を与えうる因子に関して統計解析を行った．【結果】RA-TKAの症例数は減少し，TKA患者の術前の血液検査で炎症所見は有意に低下傾向であった．術後平均14年のRA-TKAのKSSのスコアは，当院にて施行された変形性膝関節症に対するTKAの術後平均4年成績と同等であった．【考察】薬物療法の変遷の影響と思われるRA-TKA数の減少傾向を認めた．術後長期のKSSは比較的良好であったが，その影響因子の解明が今後の課題である．</p>

DOI： 10.5035/nishiseisai.72.212

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Language：Japanese   Publishing type：Research paper (scientific journal)  

Language：English   Publishing type：Research paper (scientific journal)  

DOI： 10.1002/micr.31034

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Language：Japanese   Publisher：西日本整形・災害外科学会  

【目的】当院における関節リウマチ(RA)に対する人工膝関節全置換術(TKA)の患者背景の変遷を調査し,長期患者立脚評価を行った.【対象と方法】2001年～2015年に当院で施行されたTKA数の推移を調査した.同期間に施行されたRAに対するTKA(RA-TKA)158例で術前の患者背景を解析した.同158例から現在の年齢が80歳以下である52例72膝を抽出し,Knee Society Score(KSS)を送付した.返答のあった28例38膝でKSSの各項目(膝の症状,満足度,手術に関する期待度)における点数,各項目に影響を与えうる因子に関して統計解析を行った.【結果】RA-TKAの症例数は減少し,TKA患者の術前の血液検査で炎症所見は有意に低下傾向であった.術後平均14年のRA-TKAのKSSのスコアは,当院にて施行された変形性膝関節症に対するTKAの術後平均4年成績と同等であった.【考察】薬物療法の変遷の影響と思われるRA-TKA数の減少傾向を認めた.術後長期のKSSは比較的良好であったが,その影響因子の解明が今後の課題である.(著者抄録)

Language：Japanese   Publisher：The Japanese Society for Spine Surgery and Related Research  

<p><b>Introduction: </b>The spinal instability neoplastic score (SINS) is a classification system used to diagnose neoplastic spinal instability. Several reports have also described its use as a screening tool to identify patients at risk of skeletal-related events. We investigated the efficacy of SINS to detect the risk of neurological deficit in patients with spinal metastases by assessing spinal metastatic instability before the onset of myelopathy.</p><p><b>Methods: </b>We performed surgery on 81 patients with cervical or thoracic lesions classified as metastatic spine disease between 2004 and 2019. In this cohort, spinal instability was assessed in 29/81 patients before the occurrence of myelopathy. Spinal instability was evaluated by SINS with computed tomography (CT) performed within 6 months of the neurological deficit. We defined patients with a score of 7 or higher as at-risk patients.</p><p><b>Results: </b>CT was performed at an average of 72 days before the onset of neurological deficits. The SINS were 2 no-metastases cases, 4 of less than 7 (stability), 15 of 7-12 (indeterminate instability), and 8 of 13-18 (instability). We were unable to detect 21% (6/29) patients at risk of SINS.</p><p><b>Conclusions: </b>We may not be able to detect approximately 20% at-risk patients with neurological deficits by SINS before myelopathy presents.</p>

DOI： 10.34371/jspineres.2022-0021

CiNii Research

Language：Japanese   Publishing type：Research paper (scientific journal)  

Language：English   Publishing type：Research paper (scientific journal)  

DOI： 10.1093/mr/road021

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Language：Japanese   Publisher：(一社)日本脊椎脊髄病学会  

はじめに:Spinal Instability Neoplastic Score(SINS)は転移性脊椎腫瘍の不安定性評価法であるが,骨有害事象リスク患者を検出する際のスクリーニング手段としても使用が報告されている.転移性脊椎腫瘍による脊髄症状発症例から,脊髄症状発症前のSINSを測定することにより,SINSによるリスク患者検出の有効性について検証した.対象と方法:2004年から2019年,頸胸椎部の転移性脊椎腫瘍に対する手術例81例から,脊髄症状発症前の画像評価が可能であった29例について調査した.脊髄症状発症半年以内に撮影されたCTからSINSを測定し,SINS7以上をリスク患者とした.結果:CT撮影時期は脊髄症状発症の平均72日前であった.SINSは転移なし2例,7未満(stability)4例,7以上～13未満(indeterminate instability)15例,13以上(instability)8例であり,21%(6/29)はリスク患者とならなかった.結語:脊髄症状発症前のSINSによる評価では約20%で脊髄症状発症の危険性を検出できない可能性が示唆された.(著者抄録)

Language：English   Publisher：Pathology Research and Practice  

Chondroblastoma (CB) is histologically characterized by oval to polygonal-shaped mononuclear neoplastic cells, multinucleated osteoclastic giant cells, and eosinophilic matrix with occasional calcification. Genetically, the majority of CBs harbor H3F3B p.K36M mutation. Despite the historical nomenclature, it has been reported that the matrix of CB is similar to osteoid rather than true cartilage; however, it remains unclear whether neoplastic cells in CB have the potential for osteoblastic differentiation. To clarify this issue, we immunohistochemically examined the expression of osteogenic and chondrogenic markers (SATB2, RUNX2, p63, and SOX9) as well as H3K36M mutant protein in 33 cases of CB. All 33 cases of CB were positive for H3K36M, while SATB2, RUNX2, p63, and SOX9 were expressed in 30/33 (91%), 33/33 (100%), 29/33 (88%), and 31/32 (97%) CB cases, respectively. Our immunohistochemical results suggest that neoplastic cells in CB frequently express both osteogenic and chondrogenic markers and may have an intermediate feature of osteoblastic and chondroblastic nature.

DOI： 10.1016/j.prp.2022.154239

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Language：English   Publishing type：Research paper (scientific journal)  

Language：English   Publisher：Oxford University Press  

骨軟部腫瘍の手術を受ける患者を対象に、止血作用や抗炎症作用などを有するトラネキサム酸(TXA)投与の有効性と安全性を検討するため、後方視的な傾向スコアマッチング解析を行った。2017年1月から2018年12月までに切開生検、辺縁切除、掻爬、広範切除を受けた骨軟部腫瘍の患者連続454例を対象とした。TXA投与の有無によってTXA投与群211例(平均47.6±20.7歳)とTXA非投与群243例(平均45.1±22.1歳)に分けた。患者454例のうち、切開生検は102例、辺縁切除は175例、掻爬は54例、広範切除は123例で行われた。術中出血量は、辺縁切除、広範切除ともにTXA投与群がTXA非投与群に比べて有意に少なかった(辺縁切除:17.3g vs.70.3g、P=0.045;広範切除:128.8g vs.273.1g、P=0.023)。周術期出血量および推定出血量も、広範切除ではTXA投与群でTXA非投与群に比べて有意に少なかった(周術期出血量:341.5g vs.686.5g、P=0.0039;推定出血量:320.7mL vs.550.6mL、P=0.030)。静脈血栓塞栓症は両群とも発生しなかった。以上より、TXA投与は、有害事象の発生率を上昇させずに、特に広範な切除術において安全かつ効果的に出血量を減少させることが示唆された。

Language：English   Publishing type：Research paper (scientific journal)   Publisher：Scientific Reports  

Denosumab is a game-changing drug for giant cell tumor of bone (GCTB); however, its clinical biomarker regarding tumor ossification of GCTB has not been elucidated. In this study, we investigated the relationship between Wnt/β-catenin signaling and the ossification of GCTB and evaluated whether endogenous nuclear β-catenin expression predicted denosumab-induced bone formation in GCTB. Genuine patient-derived primary GCTB tumor stromal cells exhibited osteoblastic characteristics. Identified osteoblastic markers and nuclear β-catenin translocation were significantly upregulated via differentiation induction and were inhibited by treating with Wnt signaling inhibitor, GGTI-286, or selective Rac1-LEF inhibitor, NSC23766. Furthermore, we reviewed the endogenous ossification and nuclear β-catenin translocation of 86 GCTB clinical samples and elucidated that intra-tumoral ossification was significantly associated with the nuclear translocation. Three-dimensional quantitative analyses (n = 13) of tumoral CT images have revealed that the nuclear β-catenin translocation of naïve GCTB samples was significantly involved with the denosumab-induced tumor ossification. Our findings suggest a close relationship between the nuclear β-catenin translocation and the osteoblastic differentiation of GCTB. Investigations of the nuclear β-catenin in naïve GCTB samples may provide a promising biomarker for predicting the ossification of GCTB following denosumab treatment.

DOI： 10.1038/s41598-022-17728-5

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PubMed

Language：English   Publisher：Kurume University School of Medicine  

<p><b>Summary:</b> <i><b>Background</b></i><b>:</b> Small-sized tumors tend to be resected without thorough assessment and are often managed in a sarcoma center after a malignancy is diagnosed. The lack of knowledge about the features of smallsized sarcomas may lead to unplanned resection. The features of small-sized soft tissue sarcomas were investigated by comparing them with those of small benign soft tissue tumors. </p><p><i><b>Methods</b></i><b>:</b> We included 17 soft tissue sarcoma cases (7 on the hands and feet and 10 on the limbs and trunk) with a diameter of under 2 cm. The features of small-sized sarcomas were compared to those of 39 benign soft tissue tumors with a diameter of under 2 cm and non-specific imaging findings (30 on the hands and feet and 9 on the limbs and trunk). The investigated features were age, sex, presence of pain, subjective increasing tumor size, and duration of observation. </p><p><i><b>Results</b></i><b>:</b> When we compared the tumors in the hands and feet, those <40 years of age (5/7 [71%] vs. 8/30 [27%], p=0.03) experiencing pain (7/7 [100%] vs. 13/30 [43%], p=0.007) were more common in patients with sarcomas than in patients with benign tumors. When we compared the tumors in the limbs and trunk, there was no significant difference in all investigated features. </p><p><i><b>Conclusion</b></i><b>:</b> Although clinical features were ineffective in distinguishing malignancy in most small-sized soft tissue tumors, we should pay attention to painful tumors of the hands and feet in younger patients.</p>

DOI： 10.2739/kurumemedj.ms69120015

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CiNii Research

Language：English   Publisher：eLife  

Increased intracellular iron spurs mitochondrial biogenesis and respiration to satisfy high-energy demand during osteoclast differentiation and bone-resorbing activities. Transferrin receptor 1 (Tfr1) mediates cellular iron uptake through endocytosis of iron-loaded transferrin and its expression increases during osteoclast differentiation. Nonetheless, the precise functions of Tfr1 and Tfr1-mediated iron uptake in osteoclast biology and skeletal homeostasis remain incompletely understood. To investigate the role of Tfr1 in osteoclast lineage cells in vivo and in vitro, we crossed Tfrc (encoding Tfr1)-floxed mice with Lyz2 (LysM)-Cre and Cathepsin K (Ctsk)-Cre mice to generate Tfrc conditional knockout mice in myeloid osteoclast precursors (Tfr1ΔLysM) or differentiated osteoclasts (Tfr1ΔCtsk), respectively. Skeletal phenotyping by μCT and histology unveiled a significant increase in trabecular bone mass with normal osteoclast number in long bones of 10-week-old young and 6-month-old adult female but not male Tfr1ΔLysM mice. Although high trabecular bone volume in long bones was observed in both male and female Tfr1ΔCtsk mice, this phenotype was more pronounced in female knockout mice. Consistent with this gender-dependent phenomena, estrogen-deficiency induced by ovariectomy decreased trabecular bone mass in Tfr1ΔLysM mice. Mechanistically, disruption of Tfr1 expression attenuated mitochondrial metabolism and cytoskeletal organization in mature osteoclasts in vitro by attenuating mitochondrial respiration and activation of the Src-Rac1-WAVE regulatory complex (WRC) axis, respectively, leading to decreased bone resorption with little impact on osteoclast differentiation. These results indicate that Tfr1-mediated iron uptake is specifically required for osteoclast function and is indispensable for bone remodeling in a gender-dependent manner.

DOI： 10.7554/eLife.73539

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Language：English   Publishing type：Research paper (scientific journal)  

DOI： 10.1007/s00432-022-04078-y

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Language：English   Publisher：久留米大学医学部  

小型軟部肉腫の臨床的特徴を小型良性軟部腫瘍と比較する後ろ向き研究を行った。2006～2019年に当院で手術が施行された直径2cm以下の軟部肉腫患者17例と、直径2cm以下で非特異的な画像所見を示す良性軟部組織腫瘍患者39例を対象とした。病変部位が手足の患者は軟部肉腫群7例(40歳未満71%、男性43%)、良性軟部組織腫瘍群30例(同27%、27%)、病変部位が四肢・体幹の患者はそれぞれ10例(同30%、30%)と9例(同44%、33%)であった。手足の腫瘍を比較した結果、40歳未満の患者と疼痛を経験した患者は軟部肉腫群が良性軟部組織腫瘍群よりも有意に多かった。四肢・体幹の腫瘍を比較した結果、調査したすべての特徴に有意差はみられなかった。以上から、大半の小型軟部腫瘍において臨床的特徴は悪性の鑑別に有効ではなかったが、若年患者における手足の有痛性腫瘍には注意を払う必要があることが示唆された。

Language：English   Publishing type：Research paper (scientific journal)  

DOI： 10.1093/jjco/hyac078

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Language：English   Publisher：Modern Pathology  

In rare cases, giant cell tumor of bone (GCTB) can undergo primary or secondary malignant transformation to malignant giant cell tumor of bone (MGCTB), but the details of the molecular alterations are still unclear. The present study aimed to elucidate the clinicopathologic and molecular features of MGCTBs based on immunohistochemistry, fluorescence in situ hybridization (FISH) and next generation sequencing (NGS) of nine MGCTBs (five primary and four secondary). Seven (78%) of 9 MGCTBs were immunohistochemically positive for H3.3 G34W. In two (22%) patients, although GCTB components were focally or diffusely positive for H3.3 G34W, their malignant components were entirely negative for H3.3 G34W, which was associated with heterozygous loss of H3F3A by FISH. NGS on four MGCTBs revealed pathogenic mutations in TP53 (n = 3), EZH2 (n = 1) and several other genes. Immunohistochemical analysis of the nine MGCTBs confirmed the p53 nuclear accumulation (n = 5) and loss of H3K27me3 expression (n = 3) and showed that they were mutually exclusive. In addition, four (80%) of five cases of pleomorphic or epithelioid cell-predominant MGCTBs were positive for p53, while three (75%) of four cases of spindle cell-predominant MGCTBs were negative for trimethylation at lysine 27 of histone 3 (H3K27me3). The results suggested that p53 alteration and dysfunction of histone methylation as evidenced by H3K27me3 loss may play an important role in the malignant progression of GCTB, and might contribute to the phenotype–genotype correlation in MGCTB. The combined histologic, immunohistochemical and molecular information may be helpful in part for the diagnosis of challenging cases.

DOI： 10.1038/s41379-021-00972-x

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Language：English   Publishing type：Research paper (scientific journal)  

Language：English   Publisher：Journal of Clinical Medicine  

Periosteal chondrosarcoma is an extremely rare malignant cartilage-forming tumour that originates from the periosteum and occurs on the surface of bone. Often, it is difficult to distinguish periosteal chondrosarcoma from other tumours, and reports in the literature are scarce. This study aims to investigate the characteristics of periosteal chondrosarcoma, focusing particularly on medullary invasion. Among 33 periosteal cartilaginous tumours, seven patients with pathologically proven periosteal chondrosarcoma were identified retrospectively. The average tumour size was 5.4 cm in the long axis; two tumours were smaller than 3.0 cm. Six tumours were resected with a wide margin, and the remaining tumour had a marginal margin. Histology revealed that six tumours (85.7%) had invaded the medullary cavity; three of these did not show invasion into the medullary cavity on MRI evaluation. Neither local recurrence nor metastasis was observed among these patients. The frequency of invasion of the medullary cavity was higher than that reported previously. The recommended treatment for periosteal chondrosarcoma is resection with an adequate margin. Therefore, surgeons should consider the possibility of medullary invasion when attempting to achieve a histologically negative margin, even if the tumour does not show invasion into the medullary cavity on MRI.

DOI： 10.3390/jcm11072062

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Language：Japanese   Publisher：西日本整形・災害外科学会  

<p>【目的】関節リウマチ（以下RA）患者において，Sauvé-Kapandji法（以下，SK法）術後3年以上経過後のX線学的変化を検討すること．【対象と方法】2007年から2019年にSK法を施行したRA17例18手の内3年以上の経過観察をし得た6例6手である．全例女性，手術時平均年齢は60歳（45-70歳），平均経過観察期間は100か月（36-160か月）であった．術後1週，最終経過観察時の単純X線を用いてCarpal height ratio（CHR），Palmar carpal subluxation ratio（PCSR），Ulnar translation index（UTI）の各値について術後1週を1とし変化率を評価した．また，この6例を含む17例18手について，術前と術後1週にて術前を1とし変化率の評価を行った．統計学的検討にはWilcoxon検定を用いて5%未満を有意差ありとした．【結果】術後1週と最終経過ではCHRは－1%，PCSRは＋7%，UTIは＋23%であり，UTIにて有意差を認めた（p＝0.0028）．また術前と術後1週の比較ではCHRは－4%，PCSRは＋11%，UTIは±0%であり，PCSRのみ有意差を認めた．【考察】RAにおけるSK法では3年以上の経過例で，術直後に対して尺側偏位が進行することが示唆された．</p>

DOI： 10.5035/nishiseisai.71.29

CiNii Research

Language：English   Publisher：Oncology (Switzerland)  

Introduction: Radiation-associated sarcoma (RAS) is one of the most life-threatening complications associated with the treatment of malignant neoplasms. Because all RAS patients have a history of radiotherapy, there have been no effective treatment options when RAS is not completely resected. Methods: We retrospectively reviewed 20 RAS patients, including 4 unresectable cases treated by carbon ion radiotherapy (CIRT). Results: The primary diseases targeted by radiotherapy included malignant lymphoma (n = 4), cervical cancer (n = 3), pharyngeal cancer (n = 3), breast cancer (n = 2), lung cancer (n = 1), rectal cancer (n = 1), maxillary cancer (n = 1), synovial sarcoma (n = 1), and benign neoplasms (n = 4). The histological diagnoses of RAS included osteosarcoma (n = 8), leiomyosarcoma (n = 3), undifferentiated pleomorphic sarcoma (n = 3), rhabdomyosarcoma (n = 1), angiosarcoma (n = 1), malignant peripheral nerve sheath tumor (n = 1), spindle cell sarcoma NOS (n = 1), and sarcoma not further specified (n = 2). The median survival time from the diagnosis of RAS was 26 months. Eleven patients underwent surgery. Five of these patients achieved a continuous disease free (CDF) status or showed no evidence disease. Four patients underwent CIRT. One of these patients with leiomyosarcoma achieved a CDF status, and the other patient with osteosarcoma achieved a partial response. On the other hand, 2 patients experienced grade 3 toxicities that required surgical treatment. Conclusion: RAS originates from various types of diseases that are treated by radiotherapy and shows diverse pathological features. Complete resection achieves a good prognosis. CIRT can be an effective and feasible option for unresectable RAS.

DOI： 10.1159/000521504

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Publisher：Gan to kagaku ryoho. Cancer &amp; chemotherapy  

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Publisher：金原出版  

DOI： 10.18888/se.0000002057

CiNii Research

Language：Japanese   Publishing type：Research paper (scientific journal)  

Language：Japanese   Publishing type：Research paper (scientific journal)  

Language：Japanese  

PubMed

Language：Japanese   Publisher：西日本整形・災害外科学会  

【目的】関節リウマチ(以下RA)患者において,Sauve-Kapandji法(以下,SK法)術後3年以上経過後のX線学的変化を検討すること.【対象と方法】2007年から2019年にSK法を施行したRA17例18手の内3年以上の経過観察をし得た6例6手である.全例女性,手術時平均年齢は60歳(45-70歳),平均経過観察期間は100ヵ月(36-160ヵ月)であった.術後1週,最終経過観察時の単純X線を用いてCarpal height ratio(CHR),Palmar carpal subluxation ratio(PCSR),Ulnar translation index(UTI)の各値について術後1週を1とし変化率を評価した.また,この6例を含む17例18手について,術前と術後1週にて術前を1とし変化率の評価を行った.統計学的検討にはWilcoxon検定を用いて5%未満を有意差ありとした.【結果】術後1週と最終経過ではCHRは-1%,PCSRは+7%,UTIは+23%であり,UTIにて有意差を認めた(p=0.0028).また術前と術後1週の比較ではCHRは-4%,PCSRは+11%,UTIは±0%であり,PCSRのみ有意差を認めた.【考察】RAにおけるSK法では3年以上の経過例で,術直後に対して尺側偏位が進行することが示唆された.(著者抄録)

Language：English   Publishing type：Research paper (scientific journal)  

DOI： 10.1038/s41416-021-01695-1

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DOI： 10.3390/jcm10214972

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DOI： https://doi.org/10.1093/jjco/hyab083

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DOI： 10.4049/jimmunol.2000783

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DOI： 10.1007/s00432-021-03616-4

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DOI： 10.1177/2192568220982571

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Language：English   Publishing type：Research paper (scientific journal)  

DOI： 10.1038/s41598-020-77898-y

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DOI： 10.1007/s00223-015-9967-7

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DOI： 10.1007/s00223-014-9883-2

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DOI： 10.1074/jbc.M113.478750

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DOI： 10.1186/1475-2867-13-21

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DOI： 10.1038/bjc.2012.579

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Country：Japan  

Event date： 2021.9

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2021.9

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2021.9

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2021.9

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2021.7

Language：Japanese   Presentation type：Oral presentation (general)  

Venue：広島   Country：Japan  

Event date： 2021.7

Language：Japanese   Presentation type：Oral presentation (general)  

Venue：広島   Country：Japan  

Event date： 2021.7

Language：Japanese   Presentation type：Oral presentation (general)  

Venue：広島   Country：Japan  

Event date： 2021.7

Language：Japanese   Presentation type：Oral presentation (general)  

Venue：広島   Country：Japan  

Event date： 2021.7

Language：Japanese   Presentation type：Oral presentation (general)  

Venue：広島   Country：Japan  

Event date： 2021.7

Language：Japanese   Presentation type：Oral presentation (general)  

Venue：広島   Country：Japan  

Event date： 2021.7

Language：Japanese   Presentation type：Oral presentation (general)  

Venue：広島   Country：Japan  

Event date： 2021.5

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2021.5

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2021.5

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2021.5

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2021.4

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2021.4

Language：Japanese   Presentation type：Oral presentation (invited, special)  

Country：Japan  

Event date： 2021.4

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2021.4

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2021.3

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2021.3

Language：Japanese  

Country：Japan  

Event date： 2021.3

Language：Japanese  

Country：Japan  

Event date： 2021.3

Language：Japanese  

Country：Japan  

Event date： 2021.3

Language：Japanese  

Country：Japan  

Event date： 2021.3

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2021.3

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2021.3

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2021.3

Language：Japanese  

Country：Japan  

Event date： 2021.3

Language：Japanese  

Country：Japan  

Event date： 2021.3

Language：Japanese  

Country：Japan  

Event date： 2021.3

Language：Japanese  

Country：Japan  

Event date： 2021.3

Language：Japanese  

Country：Japan  

Event date： 2021.3

Language：Japanese  

Country：Japan  

Event date： 2021.3

Language：Japanese  

Country：Japan  

Event date： 2019.11 - 2019.12

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2019.11 - 2019.12

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2019.11 - 2019.12

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2019.11

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2019.11

Language：Japanese   Presentation type：Symposium, workshop panel (public)  

Country：Japan  

Event date： 2019.10

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2019.10

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2019.9

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2019.9

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2019.7

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2019.7

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2019.7

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2019.7

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2019.7

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2019.7

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2019.7

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2019.7

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2019.7

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2019.6

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2019.6

Language：Japanese   Presentation type：Symposium, workshop panel (public)  

Country：Japan  

Event date： 2019.6

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2019.6

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2019.3

Language：Japanese  

Venue：アクロス福岡   Country：Japan  

Event date： 2019.3

Language：Japanese   Presentation type：Oral presentation (general)  

Venue：アクロス福岡   Country：Japan  

Event date： 2018.11

Language：Japanese   Presentation type：Oral presentation (general)  

Venue：福岡市   Country：Japan  

Event date： 2018.11

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2018.11

Language：Japanese   Presentation type：Oral presentation (general)  

Venue：岡山市   Country：Japan  

Event date： 2018.9

Language：Japanese   Presentation type：Symposium, workshop panel (public)  

Country：Japan  

Event date： 2018.8

Language：Japanese   Presentation type：Oral presentation (general)  

Venue：福岡市   Country：Japan  

Event date： 2018.7

Language：Japanese   Presentation type：Oral presentation (general)  

Venue：長崎市   Country：Japan  

LIS1の破骨細胞分化制御機構をin vitroとin vivoで解明した。

Event date： 2018.6

Language：Japanese   Presentation type：Oral presentation (general)  

Venue：福岡市   Country：Japan  

Event date： 2018.6

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

破骨細胞においてPLEKHM１ーDEF８ーRAB7複合体が骨吸収におけるライソゾーム分泌と骨代謝機構をin vivoとin vitroを用いて解明した。

Event date： 2018.6

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2018.6

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2018.5

Language：English   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2018.4

Language：English  

Country：Japan  

Event date： 2018.3

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2017.11

Language：Japanese   Presentation type：Oral presentation (general)  

Venue：米子市   Country：Japan  

Event date： 2017.9

Language：English   Presentation type：Oral presentation (general)  

Country：United States  

Event date： 2017.9

Language：Japanese   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2017.7

Language：Japanese   Presentation type：Oral presentation (general)  

Venue：福岡市   Country：Japan  

Event date： 2017.4

Language：English   Presentation type：Oral presentation (general)  

Country：Japan  

Event date： 2016.10

Language：Japanese  

Country：Japan  

Event date： 2015.10

Language：English   Presentation type：Oral presentation (general)  

Country：United States  

Event date： 2015.10

Language：English   Presentation type：Oral presentation (general)  

Country：United States  

Event date： 2014.9

Language：English  

Country：United States  

Event date： 2014.9

Language：English  

Country：United States  

Event date： 2014.9

Language：English  

Country：United States  

Event date： 2014.9

Language：English  

Country：United States  

Event date： 2013.10

Language：English  

Country：United States  

Language：Japanese  

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Language：Japanese   Publisher：(株)日本臨床社  

Language：Japanese   Publishing type：Article, review, commentary, editorial, etc. (scientific journal)  

Language：Japanese   Publisher：(公社)日本整形外科学会  

悪性軟部腫瘍に対する薬物療法は,周術期化学療法として行う場合と,切除不能・進行病変に対して行う場合に大別される.非円形細胞肉腫では,AI(adriamycin,ifosfamide)療法が周術期化学療法の標準治療であり,高悪性度腫瘍に対して実施が検討される.軟部肉腫に対する周術期化学療法は,メタ解析により,生存期間の有意な延長が示されている.わが国ではJCOG0304試験にて,良好な治療成績が示されており,これが事実上の標準治療となっている.進行例に対する一次治療の第1選択はアドリアマイシンを含むレジメンであり,ガイドライン等で推奨されている.二次治療では,トラベクテジン,エリブリン,パゾパニブや,GD(gemcitabine,docetaxel)療法が治療選択肢となるが,各国のガイドラインを参照しても,どの薬物を優先して用いるべきか明確にされていない.現在,進行軟部肉腫を対象として,JCOG1802試験が行われており,進行軟部肉腫に対する二次治療について,3剤の使い分けに関する貴重なデータが得られることが期待される.(著者抄録)

Language：Japanese   Publishing type：Article, review, commentary, editorial, etc. (scientific journal)  

Language：Japanese   Publishing type：Article, review, commentary, editorial, etc. (scientific journal)  

Language：Japanese   Publisher：(株)癌と化学療法社  

Language：Japanese   Publisher：金原出版(株)  

＜文献概要＞進行軟部肉腫に対する化学療法の一次治療で頻用されるドキソルビシン(アドリアマイシン)には不可逆的累積毒性である心毒性があり,総投与量が上限に近づいた場合や,抵抗性を示す症例ではセカンドライン化学療法が必要となる。トラベクテジン,エリブリン,パゾパニブは2010年代に悪性軟部腫瘍を適応症として薬事承認された比較的新しい薬剤であり,それらに加え,GD療法(ゲムシタビン+ドセタキセル併用療法,悪性軟部腫瘍については薬事未承認)がセカンドライン化学療法の治療選択肢となる。実臨床におけるそれぞれの薬剤の治療成績や副作用について徐々に明らかになってきたとはいえ,それらを直接比較したランダム化比較試験(RCT)は存在せず,どの薬剤を選択すべきかに関する明確なエビデンスは乏しい状況である。現在行われているJCOG1802試験により,薬剤選択において参考となる比較データが得られることが期待されている。

Language：Japanese   Publishing type：Article, review, commentary, editorial, etc. (scientific journal)  

Language：Japanese   Publishing type：Article, review, commentary, editorial, etc. (scientific journal)  

Language：Japanese   Publishing type：Article, review, commentary, editorial, etc. (scientific journal)  

Language：Japanese  

Language：English