{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,1]],"date-time":"2026-08-01T11:31:55Z","timestamp":1785583915524,"version":"3.56.0"},"reference-count":16,"publisher":"Springer Science and Business Media LLC","issue":"6","license":[{"start":{"date-parts":[[2025,5,6]],"date-time":"2025-05-06T00:00:00Z","timestamp":1746489600000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2025,5,6]],"date-time":"2025-05-06T00:00:00Z","timestamp":1746489600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/100009619","name":"Japan Agency for Medical Research and Development","doi-asserted-by":"crossref","award":["JP24ek0510046"],"award-info":[{"award-number":["JP24ek0510046"]}],"id":[{"id":"10.13039\/100009619","id-type":"DOI","asserted-by":"crossref"}]},{"name":"JSPS KAKENHI","award":["JP24K11515"],"award-info":[{"award-number":["JP24K11515"]}]}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Ann Hematol"],"published-print":{"date-parts":[[2025,6]]},"abstract":"<jats:title>Abstract<\/jats:title>\n          <jats:p>\n            <jats:italic>TP53<\/jats:italic>-mutated myelodysplastic syndrome (MDS) and acute erythroid leukemia (AEL) with complex karyotype have a very poor prognosis. The upregulation of the JAK-STAT pathway has been implicated in their pathogenesis, and inhibition of this pathway has shown promising disease control in preclinical models. Here, we report a case of refractory multi-hit <jats:italic>TP53<\/jats:italic>-mutated MDS with erythroid predominance on the verge of transitioning to AEL, which achieved hematological complete remission following allogeneic stem cell transplantation and ruxolitinib initiation. The patient exhibited chemoresistance to multiple regimens, including cytarabine with daunorubicin, high-dose cytarabine, and venetoclax with azacitidine. Despite the presence of residual disease post-transplant, complete remission was achieved two months after ruxolitinib initiation and tacrolimus tapering. At the 8-month follow-up, remission persists without evidence of relapse. This case highlights the potential of combining graft-versus-leukemia effects with ruxolitinib as a therapeutic strategy for <jats:italic>TP53<\/jats:italic>-mutated MDS\/AEL. Further studies are warranted to evaluate the efficacy and safety of this strategy in a broader clinical setting.<\/jats:p>","DOI":"10.1007\/s00277-025-06383-1","type":"journal-article","created":{"date-parts":[[2025,5,5]],"date-time":"2025-05-05T22:49:55Z","timestamp":1746485395000},"page":"3517-3522","update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Successful treatment of multi-hit TP53-mutated myelodysplastic syndromes with erythroid predominance using allogeneic stem cell transplantation and ruxolitinib"],"prefix":"10.1007","volume":"104","author":[{"given":"Seigi","family":"Oshima","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Junya","family":"Kanda","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"June","family":"Takeda","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Takashi","family":"Sakamoto","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chisaki","family":"Mizumoto","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kouhei","family":"Yamashita","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yasuhito","family":"Nannya","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Seishi","family":"Ogawa","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Akifumi","family":"Takaori-Kondo","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2025,5,6]]},"reference":[{"issue":"7","key":"6383_CR1","doi-asserted-by":"publisher","first-page":"1703","DOI":"10.1038\/s41375-022-01613-1","volume":"36","author":"JD Khoury","year":"2022","unstructured":"Khoury JD, Solary E, Abla O et al (2022) The 5th edition of the world health organization classification of haematolymphoid tumours myeloid and Histiocytic dendritic neoplasms. Leukemia 36(7):1703\u20131719. https:\/\/doi.org\/10.1038\/s41375-022-01613-1","journal-title":"Leukemia"},{"issue":"11","key":"6383_CR2","doi-asserted-by":"publisher","first-page":"1200","DOI":"10.1182\/blood.2022015850","volume":"140","author":"DA Arber","year":"2022","unstructured":"Arber DA, Orazi A, Hasserjian RP et al (2022) International consensus classification of myeloid neoplasms and acute leukemias: integrating morphologic, clinical, and genomic data. Blood 140(11):1200\u20131228. https:\/\/doi.org\/10.1182\/blood.2022015850","journal-title":"Blood"},{"issue":"20","key":"6383_CR3","doi-asserted-by":"publisher","first-page":"2391","DOI":"10.1182\/blood-2016-03-643544","volume":"127","author":"DA Arber","year":"2016","unstructured":"Arber DA, Orazi A, Hasserjian R et al (2016) The 2016 revision to the world health organization classification of myeloid neoplasms and acute leukemia. Blood 127(20):2391\u20132405. https:\/\/doi.org\/10.1182\/blood-2016-03-643544","journal-title":"Blood"},{"issue":"9","key":"6383_CR4","doi-asserted-by":"publisher","first-page":"1940","DOI":"10.1038\/leu.2013.144","volume":"27","author":"V Grossmann","year":"2013","unstructured":"Grossmann V, Bacher U, Haferlach C et al (2013) Acute erythroid leukemia (AEL) can be separated into distinct prognostic subsets based on cytogenetic and molecular genetic characteristics. Leukemia 27(9):1940\u20131943. https:\/\/doi.org\/10.1038\/leu.2013.144","journal-title":"Leukemia"},{"issue":"1","key":"6383_CR5","doi-asserted-by":"publisher","first-page":"534","DOI":"10.1186\/s12885-017-3528-6","volume":"17","author":"S Qiu","year":"2017","unstructured":"Qiu S, Jiang E, Wei H et al (2017) An analysis of 97 previously diagnosed de Novo adult acute erythroid leukemia patients following the 2016 revision to world health organization classification. BMC Cancer 17(1):534. https:\/\/doi.org\/10.1186\/s12885-017-3528-6","journal-title":"BMC Cancer"},{"issue":"10","key":"6383_CR6","doi-asserted-by":"publisher","first-page":"1985","DOI":"10.1182\/blood-2009-09-243964","volume":"115","author":"RP Hasserjian","year":"2010","unstructured":"Hasserjian RP, Zuo Z, Garcia C et al (2010) Acute erythroid leukemia: a reassessment using criteria refined in the 2008 WHO classification. Blood 115(10):1985\u20131992. https:\/\/doi.org\/10.1182\/blood-2009-09-243964","journal-title":"Blood"},{"issue":"4","key":"6383_CR7","doi-asserted-by":"publisher","first-page":"837","DOI":"10.3390\/ijms18040837","volume":"18","author":"AM Almeida","year":"2017","unstructured":"Almeida AM, Prebet T, Itzykson R et al (2017) Clinical outcomes of 217 patients with acute erythroleukemia according to treatment type and line: A retrospective multinational study. Int J Mol Sci 18(4):837. https:\/\/doi.org\/10.3390\/ijms18040837","journal-title":"Int J Mol Sci"},{"issue":"15","key":"6383_CR8","doi-asserted-by":"publisher","first-page":"3941","DOI":"10.3390\/cancers15153941","volume":"15","author":"K Gera","year":"2023","unstructured":"Gera K, Martir D, Xue W, Wingard J (2023) Survival after pure (acute) erythroid leukemia in the united States: A SEER-based study. Cancers (Basel) 15(15):3941. https:\/\/doi.org\/10.3390\/cancers15153941","journal-title":"Cancers (Basel)"},{"issue":"5","key":"6383_CR9","doi-asserted-by":"publisher","first-page":"410","DOI":"10.1158\/2643-3230","volume":"3","author":"J Takeda","year":"2022","unstructured":"Takeda J, Yoshida K, Nakagawa M et al (2022) Amplified EPOR JAK2 genes define a unique subtype of acute erythroid leukemia. 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