{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,15]],"date-time":"2026-09-15T18:50:26Z","timestamp":1789498226583,"version":"build-2803163510"},"reference-count":9,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2024,1,9]],"date-time":"2024-01-09T00:00:00Z","timestamp":1704758400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["eJHaem"],"published-print":{"date-parts":[[2024,2]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>Hairy cell leukemia (HCL) is a rare lymphoproliferative disorder classically presenting with cytopenia and recurrent infections but atypical manifestations such as bone lesions, skin lesions and effusion have been described.<\/jats:p><jats:p>We report here an unusual meningeal localization in a 33 years old man who presented with headache, hand paresthesia and visual symptoms. Brain magnetic resonance imaging revealed an occipital meningeal lesion. Diagnostic explorations led to the diagnosis of classical HCL with meningeal localization. After treatment by cladribine and rituximab the patient rapidly improved and is still in complete remission 12 months after end of treatment.<\/jats:p><jats:p>The literature review identified 9 other cases of HCL with central nervous system localization (CNS) presenting with brain parenchyma and\/or meninges localization. Four out of 9 patients presented with hyperleukocytosis. Most patients experienced good responses with various treatments. Cladribine alone or with rituximab led to complete responses similar to our patient. In our patient, molecular biology revealed KLF2 mutations, which implication in the atypical localization could be suspected but would need dedicated studies.<\/jats:p><jats:p>In conclusion, CNS localizations of HCL are rare but can be observed and treatment with cladribine alone or with rituximab appears as an effective strategy.<\/jats:p>","DOI":"10.1002\/jha2.841","type":"journal-article","created":{"date-parts":[[2024,1,10]],"date-time":"2024-01-10T06:54:20Z","timestamp":1704869660000},"page":"242-246","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":4,"title":["Atypical meningeal localization of classical hairy cell leukemia with an impressive response to rituximab and cladribine association. A case report and literature review"],"prefix":"10.1002","volume":"5","author":[{"ORCID":"https:\/\/orcid.org\/0009-0002-6468-0001","authenticated-orcid":false,"given":"Fabien","family":"Claves","sequence":"first","affiliation":[{"name":"Hematology Department University Hospital of Grenoble Alpes  Grenoble France"},{"name":"Grenoble Alpes University  Grenoble France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sylvain","family":"Carras","sequence":"additional","affiliation":[{"name":"Hematology Department University Hospital of Grenoble Alpes  Grenoble France"},{"name":"Grenoble Alpes University  Grenoble France"},{"name":"Genetic of Hematological Malignancies Department University Hospital of Grenoble  Grenoble France"},{"name":"Institute for Advanced Biosciences  Grenoble France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Barbara","family":"Burroni","sequence":"additional","affiliation":[{"name":"Pathology Department Cochin University Hospital  Paris France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5967-8225","authenticated-orcid":false,"given":"Elsa","family":"Maitre","sequence":"additional","affiliation":[{"name":"University Hospital Caen Caen France"},{"name":"INSERM U1245 Normandie University  Caen France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jean","family":"Boutonnat","sequence":"additional","affiliation":[{"name":"Pathology Department University Hospital of Grenoble Alpes  Grenoble France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6863-9992","authenticated-orcid":false,"given":"Xavier","family":"Troussard","sequence":"additional","affiliation":[{"name":"University Hospital Caen Caen France"},{"name":"INSERM U1245 Normandie University  Caen France"},{"name":"Hematology Department University Hospital Caen Caen France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lysiane","family":"Molina","sequence":"additional","affiliation":[{"name":"Hematology Department University Hospital of Grenoble Alpes  Grenoble France"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2024,1,9]]},"reference":[{"key":"e_1_2_9_2_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajh.26390"},{"key":"e_1_2_9_3_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41375-022-01764-1"},{"key":"e_1_2_9_4_1","doi-asserted-by":"publisher","DOI":"10.1111\/bjh.12908"},{"key":"e_1_2_9_5_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41408-020-0328-z"},{"key":"e_1_2_9_6_1","doi-asserted-by":"publisher","DOI":"10.5045\/br.2020.2020069"},{"key":"e_1_2_9_7_1","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2019-121826"},{"key":"e_1_2_9_8_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00277-020-04349-z"},{"key":"e_1_2_9_9_1","doi-asserted-by":"publisher","DOI":"10.3390\/cancers14081904"},{"key":"e_1_2_9_10_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.1013168108"}],"container-title":["eJHaem"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/jha2.841","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,2,23]],"date-time":"2024-02-23T16:49:14Z","timestamp":1708706954000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/jha2.841"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,1,9]]},"references-count":9,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2024,2]]}},"alternative-id":["10.1002\/jha2.841"],"URL":"https:\/\/doi.org\/10.1002\/jha2.841","archive":["Portico"],"relation":{},"ISSN":["2688-6146","2688-6146"],"issn-type":[{"value":"2688-6146","type":"print"},{"value":"2688-6146","type":"electronic"}],"subject":[],"published":{"date-parts":[[2024,1,9]]},"assertion":[{"value":"2023-10-01","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2023-12-18","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2024-01-09","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}