{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,27]],"date-time":"2026-08-27T11:04:10Z","timestamp":1787828650944,"version":"build-2784847793"},"reference-count":37,"publisher":"Wiley","issue":"2","license":[{"start":{"date-parts":[[2018,11,11]],"date-time":"2018-11-11T00:00:00Z","timestamp":1541894400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100002973","name":"Daiichi Sankyo","doi-asserted-by":"crossref","id":[{"id":"10.13039\/501100002973","id-type":"DOI","asserted-by":"crossref"}]},{"name":"Leiden University Medical Center (LUMC)"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Histopathology"],"published-print":{"date-parts":[[2019,1]]},"abstract":"Aims<\/jats:title>Localised\u2010 and diffuse\u2010type tenosynovial giant cell tumours (TGCT<\/jats:styled-content>) are regarded as different clinical and radiological TGCT<\/jats:styled-content> types. However, genetically and histopathologically they seem indistinguishable. We aimed to correlate CSF<\/jats:styled-content>1<\/jats:italic> expression and CSF<\/jats:styled-content>1<\/jats:italic> rearrangement with the biological behaviour of different TGCT<\/jats:styled-content>\u2010types with clinical outcome (recurrence).<\/jats:p><\/jats:sec>Methods and results<\/jats:title>Along a continuum of extremes, therapy\u2010naive knee TGCT<\/jats:styled-content> patients with >3\u2010year follow\u2010up, mean age 43 (range\u00a0=\u00a06\u201371) years and 56% females were selected. Nine localised (two recurrences), 16 diffuse\u2010type (nine recurrences) and four synovitis as control were included. Rearrangement of the CSF<\/jats:styled-content>1<\/jats:italic> locus was evaluated with split\u2010apart fluorescence in\u2010situ<\/jats:italic> hybridisation (FISH<\/jats:styled-content>) probes. Regions were selected to score after identifying CSF<\/jats:styled-content>1<\/jats:italic>\u2010expressing regions, using mRNA ISH<\/jats:styled-content> with the help of digital correlative microscopy. CSF<\/jats:styled-content>1<\/jats:italic> rearrangement was considered positive in samples containing >2 split signals\/100 nuclei. Irrespective of TGCT<\/jats:styled-content>\u2010subtype, all cases showed CSF<\/jats:styled-content>1<\/jats:italic> expression and in 76% CSF<\/jats:styled-content>1<\/jats:italic> rearrangement was detected. Quantification of CSF<\/jats:styled-content>1<\/jats:italic>\u2010expressing cells was not informative, due to the extensive intratumour heterogeneity. Of the four synovitis cases, two also showed CSF<\/jats:styled-content>1<\/jats:italic> expression without CSF<\/jats:styled-content>1<\/jats:italic> rearrangement. No correlation between CSF<\/jats:styled-content>1<\/jats:italic> expression or rearrangement with clinical subtype and local recurrence was detected. Both localised and diffuse TGCT<\/jats:styled-content> cases showed a scattered distribution in the tissue of CSF<\/jats:styled-content>1<\/jats:italic>\u2010expressing cells.<\/jats:p><\/jats:sec>Conclusion<\/jats:title>In diagnosing TGCT<\/jats:styled-content>, CSF<\/jats:styled-content>1 <\/jats:italic>mRNA<\/jats:styled-content>\u2010ISH<\/jats:styled-content>, in combination with CSF<\/jats:styled-content>1<\/jats:italic> split\u2010apart FISH<\/jats:styled-content> using digital correlative microscopy, is an auxiliary diagnostic tool to identify rarely occurring neoplastic cells. This combined approach allowed us to detect CSF<\/jats:styled-content>1<\/jats:italic> rearrangement in 76% of the TGCT<\/jats:styled-content> cases. Neither CSF<\/jats:styled-content>1<\/jats:italic> expression nor presence of CSF<\/jats:styled-content>1<\/jats:italic> rearrangement could be associated with the difference in biological behaviour of TGCT<\/jats:styled-content>.<\/jats:p><\/jats:sec>","DOI":"10.1111\/his.13744","type":"journal-article","created":{"date-parts":[[2018,8,30]],"date-time":"2018-08-30T04:13:51Z","timestamp":1535602431000},"page":"332-340","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":42,"title":["Does CSF<\/scp>1<\/i> overexpression or rearrangement influence\u00a0biological behaviour in tenosynovial giant cell\u00a0tumours of the knee?"],"prefix":"10.1111","volume":"74","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-8980-3955","authenticated-orcid":false,"given":"Monique J L","family":"Mastboom","sequence":"first","affiliation":[{"name":"Department of Orthopaedic Surgery Leiden University Medical Center Leiden the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Daisy M","family":"Hoek","sequence":"additional","affiliation":[{"name":"Department of Cell and Chemical biology Leiden University Medical Center Leiden the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Judith V M G","family":"Bov\u00e9e","sequence":"additional","affiliation":[{"name":"Department of Pathology Leiden University Medical Center Leiden the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michiel A J","family":"van de Sande","sequence":"additional","affiliation":[{"name":"Department of Orthopaedic Surgery Leiden University Medical Center Leiden the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1228-4245","authenticated-orcid":false,"given":"K\u00e1roly","family":"Szuhai","sequence":"additional","affiliation":[{"name":"Department of Cell and Chemical biology Leiden University Medical Center Leiden the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2018,11,11]]},"reference":[{"key":"e_1_2_7_2_1","first-page":"100","volume-title":"WHO classification of tumours of soft tissue and bone","author":"St Aubain S","year":"2013"},{"key":"e_1_2_7_3_1","first-page":"102","volume-title":"WHO classification of tumours of soft tissue and bone","author":"St Aubain S","year":"2013"},{"key":"e_1_2_7_4_1","doi-asserted-by":"publisher","DOI":"10.1080\/17453674.2017.1361126"},{"key":"e_1_2_7_5_1","first-page":"731","article-title":"Pigmented villonodular synovitis, bursitis and tenosynovitis","volume":"31","author":"Jaffe HLLL","year":"1941","journal-title":"Arch. 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