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In the era before the introduction of tyrosine kinase inhibitors (TKIs), the only potentially curative treatment was allogeneic hematopoietic stem cell transplantation (HSCT). Here, we present the case of a patient affected by CML who experienced a relapse 20\u00a0years after allogeneic HSCT. Following relapse, the patient was treated with imatinib and bosutinib, resulting in a deep molecular response and successfully discontinued treatment. Additional analysis including whole-exome sequencing and RNA sequencing provided some insights on the molecular mechanisms of the relapse: the identification of the fusion transcript <jats:italic>KANSL1::ARL17A<\/jats:italic> (<jats:italic>KANSARL)<\/jats:italic>, a cancer predisposition fusion gene, could justify a condition of genomic instability which may be associated with the onset and\/or probably the late relapse of his CML.<\/jats:p>","DOI":"10.1007\/s00277-024-05649-4","type":"journal-article","created":{"date-parts":[[2024,2,7]],"date-time":"2024-02-07T03:01:51Z","timestamp":1707274911000},"page":"1561-1568","update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":5,"title":["Late relapse of chronic myeloid leukemia after allogeneic bone marrow transplantation points to KANSARL (KANSL1::ARL17A) alteration: a case report with insights on the molecular landscape"],"prefix":"10.1007","volume":"103","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-1633-6310","authenticated-orcid":false,"given":"Diletta","family":"Fontana","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Giovanni Paolo Maria","family":"Zambrotta","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Antonio","family":"Scannella","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rocco","family":"Piazza","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Carlo","family":"Gambacorti-Passerini","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2024,2,7]]},"reference":[{"issue":"14","key":"5649_CR1","doi-asserted-by":"publisher","first-page":"1031","DOI":"10.1056\/nejm200104053441401","volume":"344","author":"BJ Druker","year":"2001","unstructured":"Druker BJ, Talpaz M, Resta DJ, Peng B, Buchdunger E, Ford JM et al (2001) Efficacy and safety of a specific inhibitor of the BCR-ABL tyrosine kinase in chronic myeloid leukemia. 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