{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,9]],"date-time":"2026-10-09T19:24:37Z","timestamp":1791573877573,"version":"4.3.4"},"reference-count":36,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2020,11,24]],"date-time":"2020-11-24T00:00:00Z","timestamp":1606176000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/501100004663","name":"Ministry of Science and Technology, Taiwan","doi-asserted-by":"publisher","award":["108\u20102320\u2010B\u2010182A\u2010017\u2010MY3"],"award-info":[{"award-number":["108\u20102320\u2010B\u2010182A\u2010017\u2010MY3"]}],"id":[{"id":"10.13039\/501100004663","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Histopathology"],"published-print":{"date-parts":[[2021,4]]},"abstract":"<jats:sec><jats:title>Aims<\/jats:title><jats:p><jats:italic>USP6<\/jats:italic> rearrangement underpins self\u2010limiting fibroblastic\/myofibroblastic neoplasms, including nodular fasciitis (NF), myositis ossificans (MO), aneurysmal bone cyst (ABC), and related variants. The aim of this study was to characterise <jats:italic>UPS6<\/jats:italic> and fusion partners in order to delineate the clinicopathological, genetic and bone\u2010forming features in such lesions of soft tissue (ST).<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods and results<\/jats:title><jats:p>Break\u2010apart fluorescence <jats:italic>in\u2010situ<\/jats:italic> hybridisation (FISH) validated <jats:italic>USP6<\/jats:italic> rearrangement in 31 of 35 NF [comprising three of three fasciitis ossificans (FO) cases, seven of eight cellular variant of fibroma of tendon sheath (C\u2010FTS), four of six MO, three of three ST\u2010ABC, and two of two fibro\u2010osseous pseudotumours of digits (FOPD)]. As determined with FISH and reverse transcription polymerase chain reaction, <jats:italic>MYH9<\/jats:italic>\u2013<jats:italic>USP6<\/jats:italic> was the commonest fusion in four C\u2010FTS and 20 NF, including one intravascular case and two infantile (one retroperitoneal) cases. The presence of <jats:italic>MYH9<\/jats:italic>\u2013<jats:italic>USP6<\/jats:italic> confirmed the diagnosis of two NFs&gt;\u00a050\u00a0mm with prominent ischaemic necrosis. <jats:italic>COL1A1<\/jats:italic>\u2013<jats:italic>USP6<\/jats:italic> was predominant in ossifying lesions, including all FO, MO, ST\u2010ABC and FOPD with identified partner genes, and was also present in non\u2010ossifying head and neck NF (HN\u2010NF) and C\u2010FTS in two cases each. A cervical NF of a 14\u2010month\u2010old girl harboured the novel <jats:italic>COL1A2<\/jats:italic>\u2013<jats:italic>USP6<\/jats:italic>. Ossifying lesions showed considerable genetic and morphological overlaps. Sharing <jats:italic>COL1A1<\/jats:italic>\u2013<jats:italic>USP6<\/jats:italic>, FO and FOPD showed similar central or haphazard bone matrix deposition. Besides zonation of outward bone maturation, four <jats:italic>COL1A1<\/jats:italic>\u2013<jats:italic>USP6<\/jats:italic>\u2010positive MO had incipient to sieve\u2010like pseudocysts reminiscent of ST\u2010ABC.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p><jats:italic>MYH9<\/jats:italic>\u2013<jats:italic>USP6<\/jats:italic> is present in some C\u2010FTS and most NF, including rare variants, but is unrelated to bone formation. All bone\u2010forming <jats:italic>USP6<\/jats:italic>\u2010rearranged lesions adopt <jats:italic>COL1A1<\/jats:italic> as the 5\u2032 partner, indicating close genetic kinships. However, <jats:italic>COL1A1<\/jats:italic>\/<jats:italic>COL1A2<\/jats:italic> also contributes to the pathogenesis of minor subsets of non\u2010ossifying <jats:italic>USP6<\/jats:italic>\u2010rearranged HN\u2010NF and C\u2010FTS.<\/jats:p><\/jats:sec>","DOI":"10.1111\/his.14268","type":"journal-article","created":{"date-parts":[[2020,10,1]],"date-time":"2020-10-01T05:24:41Z","timestamp":1601529881000},"page":"676-689","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":25,"title":["Clinicopathological and molecular characterisation of <i>USP6<\/i>\u2010rearranged soft tissue neoplasms: the evidence of genetic relatedness indicates an expanding family with variable bone\u2010forming capacity"],"prefix":"10.1111","volume":"78","author":[{"given":"Jui\u2010Chu","family":"Wang","sequence":"first","affiliation":[{"name":"Department of Anatomical Pathology Kaohsiung Chang Gung Memorial Hospital and Chang Gung University College of Medicine Kaohsiung Taiwan"},{"name":"Department of Medical Laboratory Science and Biotechnology School of Medical and Health Sciences Fooyin University Kaohsiung Taiwan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9403-2489","authenticated-orcid":false,"given":"Wan\u2010Shan","family":"Li","sequence":"additional","affiliation":[{"name":"Department of Pathology Chi\u2010Mei Medical Centre Tainan Taiwan"},{"name":"Department of Medical Laboratory Science and Biotechnology Chung Hwa University of Medical Technology Tainan Taiwan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yu\u2010Chien","family":"Kao","sequence":"additional","affiliation":[{"name":"Department of Pathology Shuang Ho Hospital Taipei Medical University Taipei Taiwan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jen\u2010Chieh","family":"Lee","sequence":"additional","affiliation":[{"name":"Department and Graduate Institute of Pathology 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