{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,26]],"date-time":"2026-08-26T14:45:56Z","timestamp":1787755556396,"version":"build-2784847793"},"reference-count":13,"publisher":"Ovid Technologies (Wolters Kluwer Health)","issue":"1","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"abstract":"<jats:sec>\n                    <jats:title>Case:<\/jats:title>\n                    <jats:p>We report a patient with hypereosinophilia-associated massive osteolytic lesion of the sacrum who was admitted to our hospital. Genetic analysis revealed that atypical eosinophilic cells were positive for FIP1-like-1-platelet-derived growth factor receptor-alpha (FIP1L1-PDGFRA) fusion gene. Treatment was initiated with oral administration of imatinib mesylate. The patient responded rapidly to this medication with a marked reduction in eosinophilia both from the peripheral blood and FIP1L1-PDGFRA fusion gene in the bone marrow within 2 weeks, followed by gradual osteosclerotic repair of the sacrum.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions:<\/jats:title>\n                    <jats:p>This case study found that the drug imatinib proved very effective in the treatment of this rare condition.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.2106\/jbjs.cc.19.00126","type":"journal-article","created":{"date-parts":[[2020,3,20]],"date-time":"2020-03-20T06:14:20Z","timestamp":1584684860000},"page":"e0126-e0126","source":"Crossref","is-referenced-by-count":0,"title":["The Efficacy of Imatinib Mesylate in the Treatment of a Rare Lytic Bone Lesion Caused by Hypereosinophilic Syndrome\/Chronic Eosinophilic Leukemia"],"prefix":"10.2106","volume":"10","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-4917-8606","authenticated-orcid":false,"given":"Hiroshi","family":"Nomura","sequence":"first","affiliation":[{"name":"Department of Orthopaedic Surgery, Hiroshima Red Cross Hospital & Atomic-Bomb Survivors Hospital, Hiroshima, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9105-8632","authenticated-orcid":false,"given":"Koji","family":"Iwato","sequence":"additional","affiliation":[{"name":"Department of Hematology, Hiroshima Red Cross Hospital & Atomic-Bomb Survivors Hospital, Hiroshima, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Megumu","family":"Fujiwara","sequence":"additional","affiliation":[{"name":"Department of Pathology, Hiroshima Red Cross Hospital & Atomic-Bomb Survivors Hospital, Hiroshima, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4669-1575","authenticated-orcid":false,"given":"Yugo","family":"Takano","sequence":"additional","affiliation":[{"name":"Department of Orthopaedic Surgery, Hiroshima Red Cross Hospital & Atomic-Bomb Survivors Hospital, Hiroshima, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6915-353X","authenticated-orcid":false,"given":"Junichi","family":"Arima","sequence":"additional","affiliation":[{"name":"Department of Orthopaedic Surgery, Hiroshima Red Cross Hospital & Atomic-Bomb Survivors Hospital, Hiroshima, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"276","published-online":{"date-parts":[[2020]]},"reference":[{"issue":"5","key":"bib1-20230720","doi-asserted-by":"crossref","first-page":"e1","DOI":"10.1097\/MD.0b013e3182a71eba","article-title":"The spectrum of FIP1L1-PDGFRA-associated chronic eosinophilic leukemia. New Insights based on a survey of 44 cases","volume":"92","author":"Legrand","year":"2013","journal-title":"Medicine."},{"issue":"8","key":"bib2-20230720","doi-asserted-by":"crossref","first-page":"2879","DOI":"10.1182\/blood-2003-06-1824","article-title":"The FIP1L1-PDGFRA fusion tyrokinase in hypereosinophilic syndrome and chronic eosinophilic leukemia: implications for diagnosis, classification, and management","volume":"103","author":"Gotlib","year":"2004","journal-title":"Blood."},{"issue":"5","key":"bib3-20230720","doi-asserted-by":"crossref","first-page":"765","DOI":"10.1038\/sj.leu.2400634","article-title":"Hypereosinophilic syndrome or chronic eosinophilic leukemia: report of a case with a lytic bone lesion","volume":"11","author":"Juneja","year":"1997","journal-title":"Leukemia."},{"issue":"5","key":"bib4-20230720","doi-asserted-by":"crossref","first-page":"544","DOI":"10.1111\/j.1365-2141.2006.06221.x","article-title":"Chronic eosinophilic leukemia with FIP1L1-PDGFRA fusion and T6741 mutation that evolved from Langerhans cell histiocytosis with eosinophilia after chemotherapy","volume":"134","author":"Ohnishi","year":"2006","journal-title":"Br J Haematol."},{"issue":"13","key":"bib5-20230720","doi-asserted-by":"crossref","first-page":"1201","DOI":"10.1056\/NEJMoa025217","article-title":"A tyrosine kinase created by fusion of the PDGFRA and FIP1L1 genes as a therapeutic target of imatinib in idiopathic hypereosinophilic syndrome","volume":"348","author":"Cools","year":"2003","journal-title":"N Engl J Med."},{"issue":"2","key":"bib6-20230720","doi-asserted-by":"crossref","first-page":"473","DOI":"10.1182\/blood-2003-08-2798","article-title":"Molecular remission and reversal of myelofibrosis in response to imatinib mesylate treatment in patients with the myeloproliferative variant of hypereosinophilic syndrome","volume":"103","author":"Klion","year":"2004","journal-title":"Blood."},{"issue":"6","key":"bib7-20230720","doi-asserted-by":"crossref","first-page":"1220","DOI":"10.7326\/0003-4819-68-6-1220","article-title":"The hypereosinophilic syndromes","volume":"68","author":"Hardy","year":"1968","journal-title":"Ann Intern Med."},{"issue":"17","key":"bib8-20230720","doi-asserted-by":"crossref","first-page":"3842","DOI":"10.1002\/cncr.24440","article-title":"The 2008 World Health Organization classification system for myeloproliferative neoplasms: order out of chaos","volume":"115","author":"Tefferi","year":"2009","journal-title":"Cancer."},{"issue":"2","key":"bib9-20230720","doi-asserted-by":"crossref","first-page":"174","DOI":"10.1007\/s12185-010-0529-5","article-title":"Classification and diagnosis of myeloproliferative neoplasms according to the 2008 World Health Organization criteria","volume":"91","author":"Wadleigh","year":"2010","journal-title":"Int J Hematol."},{"issue":"6","key":"bib10-20230720","doi-asserted-by":"crossref","first-page":"704","DOI":"10.1182\/blood-2016-10-695973","article-title":"Myeloid neoplasms with eosinophilia","volume":"129","author":"Reiter","year":"2017","journal-title":"Blood."},{"issue":"11","key":"bib11-20230720","doi-asserted-by":"crossref","first-page":"1243","DOI":"10.1002\/ajh.24880","article-title":"World Health Organization-defined eosinophilic disorders: 2017 update on diagnosis, risk stratification, and management","volume":"92","author":"Gotlib","year":"2017","journal-title":"Am J Hematol."},{"issue":"10","key":"bib12-20230720","doi-asserted-by":"crossref","first-page":"1211","DOI":"10.5858\/arpa.2015-0199-RA","article-title":"Langerhans cell histiocytosis: a clinicopathologic review and molecular pathogenetic update","volume":"139","author":"Harmon","year":"2015","journal-title":"Arch Pathol Lab Med."},{"issue":"10","key":"bib14-20230720","doi-asserted-by":"crossref","first-page":"4071","DOI":"10.1182\/blood-2005-08-3153","article-title":"The FIP1L1-PDGFRA fusion gene cooperates with IL-5 to induce murine hypereosinophilic syndrome (HES)\/chronic eosinophilic leukemia (CEL)-like disease","volume":"107","author":"Yamada","year":"2006","journal-title":"Blood."}],"container-title":["JBJS Case Connector"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.ovid.com\/10.2106\/JBJS.CC.19.00126","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,8,26]],"date-time":"2026-08-26T13:44:45Z","timestamp":1787751885000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.ovid.com\/10.2106\/JBJS.CC.19.00126"}},"subtitle":["A Case Report"],"short-title":[],"issued":{"date-parts":[[2020]]},"references-count":13,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2020]]}},"URL":"https:\/\/doi.org\/10.2106\/jbjs.cc.19.00126","relation":{},"ISSN":["2160-3251"],"issn-type":[{"value":"2160-3251","type":"electronic"}],"subject":[],"published":{"date-parts":[[2020]]}}}