{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,14]],"date-time":"2026-08-14T06:29:58Z","timestamp":1786688998104,"version":"build-2736575974"},"reference-count":18,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2026,8,13]],"date-time":"2026-08-13T00:00:00Z","timestamp":1786579200000},"content-version":"vor","delay-in-days":224,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2026,1,1]],"date-time":"2026-01-01T00:00:00Z","timestamp":1767225600000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Case Reports in Hematology"],"published-print":{"date-parts":[[2026,1]]},"abstract":"<jats:p>\n                    Hypereosinophilia may be the presenting feature of various conditions, including myeloid\/lymphoid neoplasms with eosinophilia and tyrosine kinase gene fusions (MLN\u2010eo\u2010TK). We report the case of a 22\u2010year\u2010old male presenting with chest pain and severe cardiac involvement in the setting of marked hypereosinophilia. Laboratory findings revealed marked leukocytosis with hypereosinophilia, mild anemia, elevated NT\u2010proBNP, impaired renal function, and elevated vitamin B12 levels. Imaging showed cardiomegaly and severe mitral regurgitation. Eosinophilia persisted despite corticosteroid and antiparasitic therapy. Bone marrow analysis revealed hypercellularity with eosinophilia, and karyotyping identified a t(5, 16) chromosomal translocation. RNA sequencing revealed a novel\n                    <jats:italic>KCTD5::PDGFRB<\/jats:italic>\n                    fusion transcript, establishing the diagnosis of MLN\u2010eo\u2010TK with cardiac involvement. Imatinib (400\u2009mg daily) induced complete hematological remission within 24\u2009hours; however, severe mitral regurgitation persisted, requiring valve replacement. To our knowledge, this is the first reported case of MLN\u2010eo\u2010TK associated with a\n                    <jats:italic>KCTD5::PDGFRB<\/jats:italic>\n                    fusion. This case highlights the importance of comprehensive cytogenetic and molecular testing in unexplained hypereosinophilia, particularly in corticosteroid\u2010refractory patients, and demonstrates a rapid and marked response to imatinib.\n                  <\/jats:p>","DOI":"10.1155\/crh\/6882482","type":"journal-article","created":{"date-parts":[[2026,8,14]],"date-time":"2026-08-14T05:56:28Z","timestamp":1786686988000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["<i>KCTD5::PDGFRB<\/i>\n                    Fusion Gene Leading to Clonal Hypereosinophilia With Cardiac Involvement in a Young Adult: A Case Report"],"prefix":"10.1155","volume":"2026","author":[{"given":"L\u00e9ona","family":"de Groote","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Daphn\u00e9","family":"Papazoglou","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jade","family":"Ku","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pierre","family":"Heimann","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0145-9046","authenticated-orcid":false,"given":"Laurent","family":"Dewispelaere","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pierre","family":"Sidon","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Florence","family":"Roufosse","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anne-Laure","family":"Tr\u00e9pant","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0007-2939-3863","authenticated-orcid":false,"given":"Nicolas","family":"Cilla","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,8,13]]},"reference":[{"key":"e_1_2_12_1_2","doi-asserted-by":"publisher","DOI":"10.1111\/all.15544"},{"key":"e_1_2_12_2_2","doi-asserted-by":"publisher","DOI":"10.1002\/ajh.27287"},{"key":"e_1_2_12_3_2","doi-asserted-by":"publisher","DOI":"10.1182\/blood.2023022417"},{"key":"e_1_2_12_4_2","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2014-02-555607"},{"key":"e_1_2_12_5_2","doi-asserted-by":"publisher","DOI":"10.1038\/s41375-023-01859-3"},{"key":"e_1_2_12_6_2","doi-asserted-by":"crossref","first-page":"1042","DOI":"10.1111\/his.14097","article-title":"Screening of PDGFRB Rearrangements in Myeloid\/Lymphoid Neoplasms Using FISH and Clinicopathological Features","volume":"76","author":"Fang H.","year":"2020","journal-title":"Histopathology"},{"key":"e_1_2_12_7_2","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2002-10-3103"},{"key":"e_1_2_12_8_2","doi-asserted-by":"publisher","DOI":"10.3324\/haematol.11420"},{"key":"e_1_2_12_9_2","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2003-08-2798"},{"key":"e_1_2_12_10_2","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2006-10-050054"},{"key":"e_1_2_12_11_2","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2003-07-2393"},{"key":"e_1_2_12_12_2","doi-asserted-by":"publisher","DOI":"10.1096\/fj.201901195rrr"},{"key":"e_1_2_12_13_2","doi-asserted-by":"crossref","DOI":"10.3390\/cells9102273","article-title":"KCTD5 Regulates Cell Migration and Adhesion via Ca2+ Signaling and Rac1 Activity","volume":"9","author":"Canales J.","year":"2020","journal-title":"Cells"},{"key":"e_1_2_12_14_2","first-page":"555","article-title":"Cardiovascular Manifestations of Hypereosinophilic Syndromes","volume":"36","author":"Ogbogu P. U.","year":"2012","journal-title":"Leukemia Research"},{"key":"e_1_2_12_15_2","first-page":"164","article-title":"Eosinophilic Myocarditis: Characteristics, Treatment, and Outcomes","volume":"108","author":"Brambatti M.","year":"2022","journal-title":"Heart"},{"key":"e_1_2_12_16_2","doi-asserted-by":"publisher","DOI":"10.1186\/2045-3701-3-45"},{"key":"e_1_2_12_17_2","article-title":"KCTD5 as a Cancer Biomarker Associated With Cell Death and Therapy Sensitivity","volume":"23","author":"Shi Y. X.","year":"2023","journal-title":"BMC Cancer"},{"key":"e_1_2_12_18_2","doi-asserted-by":"publisher","DOI":"10.1038\/nature22376"}],"container-title":["Case Reports in Hematology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1155\/crh\/6882482","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1155\/crh\/6882482","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1155\/crh\/6882482","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,8,14]],"date-time":"2026-08-14T05:56:30Z","timestamp":1786686990000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1155\/crh\/6882482"}},"subtitle":[],"editor":[{"given":"Kalyani","family":"Sen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"editor"}]}],"short-title":[],"issued":{"date-parts":[[2026,1]]},"references-count":18,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2026,1]]}},"alternative-id":["10.1155\/crh\/6882482"],"URL":"https:\/\/doi.org\/10.1155\/crh\/6882482","archive":["Portico"],"relation":{},"ISSN":["2090-6560","2090-6579"],"issn-type":[{"value":"2090-6560","type":"print"},{"value":"2090-6579","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,1]]},"assertion":[{"value":"2025-10-16","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-07-17","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-08-13","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"6882482"}}