{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,17]],"date-time":"2026-08-17T14:47:43Z","timestamp":1786978063215,"version":"build-2736575974"},"reference-count":24,"publisher":"Wiley","issue":"2","license":[{"start":{"date-parts":[[2025,3,29]],"date-time":"2025-03-29T00:00:00Z","timestamp":1743206400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"funder":[{"DOI":"10.13039\/100014823","name":"VGH and UBC Hospital Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100014823","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["eJHaem"],"published-print":{"date-parts":[[2025,4]]},"abstract":"ABSTRACT<\/jats:title>\n \n Background<\/jats:title>\n Lymphocytic variant hypereosinophilic syndrome (L\u2010HES) is a rare disorder characterized by persistent eosinophilia driven by aberrant T\u2010cell populations. Diagnosis remains challenging due to the lack of standardized diagnostic criteria.<\/jats:p>\n <\/jats:sec>\n \n Methods<\/jats:title>\n We retrospectively analyzed 18 patients diagnosed with L\u2010HES between 2016 and 2023. Comprehensive flow cytometry was performed on peripheral blood samples.<\/jats:p>\n <\/jats:sec>\n \n Results<\/jats:title>\n \n Nine patients demonstrated the classic sCD3\n \u2212<\/jats:sup>\n CD4\n +<\/jats:sup>\n CD5\n +<\/jats:sup>\n CD2\n +<\/jats:sup>\n CD45RO\n +<\/jats:sup>\n CD45RA\n \u2212<\/jats:sup>\n immunophenotype, ranging from 0.6% to 70% of total lymphocytes. Two patients showed variant sCD3\n \u2212<\/jats:sup>\n CD4\n +<\/jats:sup>\n phenotypes, five had expanded (> 10%) sCD3\n +<\/jats:sup>\n CD4\n +<\/jats:sup>\n CD7\n \u2212<\/jats:sup>\n T\u2010cells, and two displayed aberrant CD8\n +<\/jats:sup>\n T\u2010LGL populations. Clonality was established in all patients with nonclassic phenotypes by molecular TCR testing or based on uniform TRBC1. We assessed a serial gating strategy to quantify the classic L\u2010HES phenotype and found this to be highly sensitive and specific with an estimated limit of detection of 0.06% of lymphocytes. Using this strategy, we identified decreased but detectable abnormal T\u2010cells in all classic phenotype patients reassessed posttreatment, down to as low as 0.3% of lymphocytes. The identification of T\u2010LGL phenotypes with eosinophilia is a novel finding.\n <\/jats:p>\n <\/jats:sec>\n \n Conclusion<\/jats:title>\n Our study highlights the diverse immunophenotypic spectrum of L\u2010HES, emphasizing the importance of comprehensive flow cytometry analysis for accurate diagnosis.<\/jats:p>\n <\/jats:sec>","DOI":"10.1002\/jha2.1109","type":"journal-article","created":{"date-parts":[[2025,3,31]],"date-time":"2025-03-31T13:51:38Z","timestamp":1743429098000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":5,"title":["Lymphocytic Variant Hypereosinophilic Syndrome: Case Series From a Tertiary Referral Center in Canada"],"prefix":"10.1002","volume":"6","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-2318-102X","authenticated-orcid":false,"given":"Xiu Qing","family":"Wang","sequence":"first","affiliation":[{"name":"Department of Pathology and Laboratory Medicine University of British Columbia Vancouver British Columbia Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kevin","family":"Shopsowitz","sequence":"additional","affiliation":[{"name":"Department of Pathology and Laboratory Medicine University of British Columbia Vancouver British Columbia Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jack","family":"Lofroth","sequence":"additional","affiliation":[{"name":"Faculty of Medicine University of British Columbia Vancouver British Columbia Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xuehai","family":"Wang","sequence":"additional","affiliation":[{"name":"Department of Pathology and Laboratory Medicine University of British Columbia Vancouver British Columbia Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Erica","family":"Peterson","sequence":"additional","affiliation":[{"name":"Division of Hematology Dalhousie University Halifax Nova Scotia Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andrew P.","family":"Weng","sequence":"additional","affiliation":[{"name":"Department of Pathology and Laboratory Medicine University of British Columbia Vancouver British Columbia Canada"},{"name":"Terry Fox Laboratory BC Cancer Agency Vancouver British Columbia Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9551-2951","authenticated-orcid":false,"given":"Luke Y. C.","family":"Chen","sequence":"additional","affiliation":[{"name":"Division of Hematology Dalhousie University Halifax Nova Scotia Canada"},{"name":"Division of Hematology University of British Columbia Vancouver British Columbia Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2025,3,29]]},"reference":[{"key":"e_1_2_8_2_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jaci.2012.02.019"},{"key":"e_1_2_8_3_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajh.27287"},{"key":"e_1_2_8_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.clim.2022.108982"},{"key":"e_1_2_8_5_1","doi-asserted-by":"publisher","DOI":"10.1182\/hem.V14.6.7884"},{"key":"e_1_2_8_6_1","doi-asserted-by":"publisher","DOI":"10.1111\/bjh.17615"},{"key":"e_1_2_8_7_1","doi-asserted-by":"publisher","DOI":"10.1093\/ajcp\/aqaa227"},{"key":"e_1_2_8_8_1","doi-asserted-by":"publisher","DOI":"10.1111\/ejh.13437"},{"key":"e_1_2_8_9_1","doi-asserted-by":"publisher","DOI":"10.1111\/ejh.12842"},{"key":"e_1_2_8_10_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jaip.2017.06.006"},{"key":"e_1_2_8_11_1","doi-asserted-by":"publisher","DOI":"10.1111\/bjh.16332"},{"key":"e_1_2_8_12_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.clim.2023.109275"},{"key":"e_1_2_8_13_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jaci.2020.08.037"},{"key":"e_1_2_8_14_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM199910073411503"},{"key":"e_1_2_8_15_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.iac.2007.07.002"},{"key":"e_1_2_8_16_1","first-page":"1086","article-title":"CD3\u2010CD4+ Lymphoid Variant of Hypereosinophilic Syndrome: Nodal and Extranodal Histopathological and Immunophenotypic Features of a Peripheral Indolent Clonal T\u2010Cell Lymphoproliferative Disorder","volume":"100","author":"Lefevre G.","year":"2015","journal-title":"Haematologica"},{"key":"e_1_2_8_17_1","doi-asserted-by":"crossref","unstructured":"C.Carpentier L.Schandene L.Dewispelaere P.Heimann E.Cogan andF.Roufosse \u201cCD3\u2010CD4+ Lymphocytic Variant Hypereosinophilic Syndrome: Diagnostic Tools Revisited \u201dThe Journal of Allergy and Clinical Immunology: In Practice \u00a09 (2021).","DOI":"10.1016\/j.jaip.2021.01.030"},{"key":"e_1_2_8_18_1","doi-asserted-by":"publisher","DOI":"10.3389\/fimmu.2020.01765"},{"key":"e_1_2_8_19_1","doi-asserted-by":"publisher","DOI":"10.1002\/cyto.b.21874"},{"key":"e_1_2_8_20_1","doi-asserted-by":"publisher","DOI":"10.1093\/ajcp\/aqy173"},{"key":"e_1_2_8_21_1","doi-asserted-by":"publisher","DOI":"10.1038\/312541a0"},{"key":"e_1_2_8_22_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.82.15.5068"},{"key":"e_1_2_8_23_1","doi-asserted-by":"publisher","DOI":"10.1038\/leu.2012.246"},{"key":"e_1_2_8_24_1","doi-asserted-by":"publisher","DOI":"10.1097\/MD.0000000000000088"},{"key":"e_1_2_8_25_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.anndiagpath.2019.151412"}],"container-title":["eJHaem"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/jha2.1109","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,4,24]],"date-time":"2025-04-24T07:56:13Z","timestamp":1745481373000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/jha2.1109"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,3,29]]},"references-count":24,"journal-issue":{"issue":"2","published-print":{"date-parts":[[2025,4]]}},"alternative-id":["10.1002\/jha2.1109"],"URL":"https:\/\/doi.org\/10.1002\/jha2.1109","archive":["Portico"],"relation":{},"ISSN":["2688-6146","2688-6146"],"issn-type":[{"value":"2688-6146","type":"print"},{"value":"2688-6146","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,3,29]]},"assertion":[{"value":"2025-01-14","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-01-21","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-03-29","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"e1109"}}