{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,27]],"date-time":"2026-05-27T15:03:10Z","timestamp":1779894190963,"version":"3.53.1"},"reference-count":25,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2026,5,20]],"date-time":"2026-05-20T00:00:00Z","timestamp":1779235200000},"content-version":"vor","delay-in-days":19,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2026,5,1]],"date-time":"2026-05-01T00:00:00Z","timestamp":1777593600000},"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":["Cancer Reports"],"published-print":{"date-parts":[[2026,5]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Langerhans cell histiocytosis (LCH) is a rare clonal neoplasm driven by activating mutations in the MAPK pathway, leading to accumulation of pathological Langerhans cells in various tissues. LCH is increasingly associated with secondary malignancies, including leukemias, lymphomas, and solid tumors. However, chronic myeloid leukemia (CML) has not previously been reported in association with LCH. Here, we describe a rare case of CML arising in the setting of active LCH in a pediatric patient.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Case<\/jats:title>\n                    <jats:p>\n                      A 15\u2010year\u2010old boy was initially diagnosed at age 11 with single\u2010system multifocal skeletal LCH. He received 12\u2009months of vinblastine and prednisolone per the LCH\u2010III protocol but failed to achieve remission and was lost to follow\u2010up. Two years later, he re\u2010presented with fever, bone pain, leukocytosis, anemia, and thrombocytosis. Bone marrow analysis was consistent with CML and PCR confirmed\n                      <jats:italic>BCR::ABL1<\/jats:italic>\n                      p210\u2010positive CML. PET and MRI demonstrated concurrent active LCH lesions. The patient was treated with imatinib and cladribine, achieving complete hematologic and molecular remission of both diseases within 7 months.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>This rare association of CML with LCH expands the recognized spectrum of LCH\u2010associated malignancies. It underscores the importance of long\u2010term surveillance in LCH patients, with particular vigilance for secondary malignancies. It also highlights the need for further research into possible biological relationships between histiocytic and myeloid neoplasms.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/cnr2.70582","type":"journal-article","created":{"date-parts":[[2026,5,21]],"date-time":"2026-05-21T00:49:29Z","timestamp":1779324569000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Langerhans Cell Histiocytosis Associated With Chronic Myeloid Leukemia: A Pediatric Case Report"],"prefix":"10.1002","volume":"9","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-2115-9910","authenticated-orcid":false,"given":"Atbin","family":"Latifi","sequence":"first","affiliation":[{"name":"School of Medicine Arak University of Medical Sciences  Arak Iran"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0001-5046-5047","authenticated-orcid":false,"given":"Sina","family":"Yousefian","sequence":"additional","affiliation":[{"name":"Student Research Committee Arak University of Medical Sciences  Arak Iran"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,5,20]]},"reference":[{"key":"e_1_2_13_2_1","doi-asserted-by":"publisher","DOI":"10.1186\/s12964-022-00917-0"},{"key":"e_1_2_13_3_1","doi-asserted-by":"publisher","DOI":"10.1182\/blood.2019000934"},{"key":"e_1_2_13_4_1","doi-asserted-by":"publisher","DOI":"10.3389\/fimmu.2023.1275085"},{"key":"e_1_2_13_5_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMra1607548"},{"key":"e_1_2_13_6_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.dib.2022.108604"},{"key":"e_1_2_13_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejca.2022.03.036"},{"key":"e_1_2_13_8_1","doi-asserted-by":"publisher","DOI":"10.1080\/10428194.2024.2404247"},{"key":"e_1_2_13_9_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.blre.2021.100825"},{"key":"e_1_2_13_10_1","doi-asserted-by":"publisher","DOI":"10.1001\/jama.2025.0220"},{"key":"e_1_2_13_11_1","doi-asserted-by":"publisher","DOI":"10.1002\/hon.2857"},{"key":"e_1_2_13_12_1","doi-asserted-by":"publisher","DOI":"10.1111\/jdv.16850"},{"key":"e_1_2_13_13_1","doi-asserted-by":"publisher","DOI":"10.1002\/cjp2.177"},{"key":"e_1_2_13_14_1","doi-asserted-by":"publisher","DOI":"10.1111\/cup.13918"},{"key":"e_1_2_13_15_1","doi-asserted-by":"publisher","DOI":"10.3389\/fonc.2022.974307"},{"key":"e_1_2_13_16_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.humpath.2017.10.017"},{"key":"e_1_2_13_17_1","doi-asserted-by":"publisher","DOI":"10.1007\/s12032-021-01462-5"},{"issue":"3","key":"e_1_2_13_18_1","first-page":"1997","article-title":"ERK\/MAPK Signalling Pathway and Tumorigenesis","volume":"19","author":"Guo Y.\u2010J.","year":"2020","journal-title":"Experimental and Therapeutic Medicine"},{"key":"e_1_2_13_19_1","doi-asserted-by":"publisher","DOI":"10.1002\/cam4.1844"},{"key":"e_1_2_13_20_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.immuni.2019.06.025"},{"key":"e_1_2_13_21_1","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1097-0215(19970328)71:1<9::AID-IJC3>3.0.CO;2-Y"},{"key":"e_1_2_13_22_1","doi-asserted-by":"publisher","DOI":"10.1093\/ajcp\/aqy050"},{"key":"e_1_2_13_23_1","doi-asserted-by":"publisher","DOI":"10.5812\/jcp\u2010153676"},{"key":"e_1_2_13_24_1","doi-asserted-by":"publisher","DOI":"10.1002\/1097-0142(19930201)71:3<865::AID-CNCR2820710334>3.0.CO;2-0"},{"issue":"1","key":"e_1_2_13_25_1","first-page":"511","article-title":"Langerhans Cell Histiocytosis With Good Response to Low\u2010Dose Imatinib: Case Report and Literature Review","volume":"16","author":"Abdulgayoom M.","year":"2023","journal-title":"Case Reports in Oncology"},{"issue":"12","key":"e_1_2_13_26_1","first-page":"1759","article-title":"Results of TETimaX Trial of Langerhans Cell Histiocytosis Treatment and Perspectives on the Role of Imatinib Mesylate in the Era of MAPK Signaling","volume":"9","author":"Montella L.","year":"2021","journal-title":"Biomedicine"}],"container-title":["Cancer Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/cnr2.70582","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1002\/cnr2.70582","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/cnr2.70582","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,5,27]],"date-time":"2026-05-27T14:03:32Z","timestamp":1779890612000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/cnr2.70582"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,5]]},"references-count":25,"journal-issue":{"issue":"5","published-print":{"date-parts":[[2026,5]]}},"alternative-id":["10.1002\/cnr2.70582"],"URL":"https:\/\/doi.org\/10.1002\/cnr2.70582","archive":["Portico"],"relation":{},"ISSN":["2573-8348","2573-8348"],"issn-type":[{"value":"2573-8348","type":"print"},{"value":"2573-8348","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,5]]},"assertion":[{"value":"2025-07-30","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-05-13","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-05-20","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"e70582"}}