{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,3]],"date-time":"2026-06-03T16:14:34Z","timestamp":1780503274730,"version":"3.54.1"},"reference-count":13,"publisher":"Springer Science and Business Media LLC","issue":"2","license":[{"start":{"date-parts":[[2024,12,9]],"date-time":"2024-12-09T00:00:00Z","timestamp":1733702400000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2024,12,9]],"date-time":"2024-12-09T00:00:00Z","timestamp":1733702400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Int J Hematol"],"published-print":{"date-parts":[[2025,2]]},"abstract":"<jats:title>Abstract<\/jats:title>\n          <jats:p>Inotuzumab ozogamicin (InO), a CD22-directed antibody conjugated to calicheamicin, has demonstrated excellent efficacy in B-cell precursor (BCP) acute lymphoblastic leukemia (ALL). It has been used for patients with relapsed or refractory BCP-ALL as a bridge to allo-HCT. Children with Down syndrome (DS) have an increased risk of BCP-ALL and higher rates of relapse and toxicity, including treatment-related mortality. Although allo-HCT is potentially curative for relapsed or refractory ALL, post-transplant leukemic relapse rates and transplant-related mortality are dismal in patients with DS-ALL, which results in less frequent use of allo-HCT in this group than in the non-DS population. Therefore, novel and less toxic therapeutic strategies are required to improve outcomes. Here we report the case of a child with DS who was diagnosed with a second relapse of BCP-ALL and has maintained complete remission through regular single-agent InO therapy. Single-agent maintenance using InO can be a good option to avoid subsequent relapse in patients with relapsed or refractory BCP-ALL who cannot proceed to allo-HCT and require less-toxic treatments.<\/jats:p>","DOI":"10.1007\/s12185-024-03890-1","type":"journal-article","created":{"date-parts":[[2024,12,9]],"date-time":"2024-12-09T02:53:29Z","timestamp":1733712809000},"page":"276-280","update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":5,"title":["Inotuzumab ozogamicin for relapse prevention in a boy with Down syndrome and relapsed acute lymphoblastic leukemia"],"prefix":"10.1007","volume":"121","author":[{"given":"Atsushi","family":"Kohso","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4727-6050","authenticated-orcid":false,"given":"Hidemi","family":"Toyoda","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ryo","family":"Hanaki","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kaori","family":"Niwa","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yosuke","family":"Okumura","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mari","family":"Morimoto","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Takahiro","family":"Ito","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Masahiro","family":"Hirayama","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2024,12,9]]},"reference":[{"key":"3890_CR1","doi-asserted-by":"publisher","first-page":"70","DOI":"10.1182\/blood-2013-06-509463","volume":"123","author":"TD Buitenkamp","year":"2014","unstructured":"Buitenkamp TD, Izraeli S, Zimmermann M, Forestier E, Heerema NA, van den Heuvel-Eibrink MM, et al. Acute lymphoblastic leukemia in children with Down syndrome: a retrospective analysis from the Ponte di Legno study group. Blood. 2014;123:70\u20137.","journal-title":"Blood"},{"key":"3890_CR2","doi-asserted-by":"publisher","first-page":"1126","DOI":"10.1002\/pbc.24918","volume":"61","author":"JK Hitzler","year":"2014","unstructured":"Hitzler JK, He W, Doyle J, Cairo M, Camitta BM, Chan KW, et al. Outcome of transplantation for acute lymphoblastic leukemia in children with Down syndrome. Pediatr Blood Cancer. 2014;61:1126\u20138.","journal-title":"Pediatr Blood Cancer"},{"key":"3890_CR3","doi-asserted-by":"publisher","first-page":"98","DOI":"10.1111\/bjh.12348","volume":"162","author":"F Meyr","year":"2013","unstructured":"Meyr F, Escherich G, Mann G, Klingebiel T, Kulozik A, Rossig C, et al. Outcomes of treatment for relapsed acute lymphoblastic leukaemia in children with Down syndrome. Br J Haematol. 2013;162:98\u2013106.","journal-title":"Br J Haematol"},{"key":"3890_CR4","doi-asserted-by":"publisher","first-page":"218","DOI":"10.1200\/JCO.23.00389","volume":"42","author":"KR Rabin","year":"2024","unstructured":"Rabin KR, Devidas M, Chen Z, Ji L, Kairalla J, Hitzler JK, et al. Outcomes in children, adolescents, and young adults with down syndrome and ALL: a report from the Children\u2019s Oncology Group. J Clin Oncol. 2024;42:218\u201327.","journal-title":"J Clin Oncol"},{"key":"3890_CR5","doi-asserted-by":"publisher","first-page":"740","DOI":"10.1056\/NEJMoa1509277","volume":"375","author":"HM Kantarjian","year":"2016","unstructured":"Kantarjian HM, DeAngelo DJ, Stelljes M, Martinelli G, Liedtke M, Stock W, et al. Inotuzumab ozogamicin versus standard therapy for acute lymphoblastic leukemia. N Engl J Med. 2016;375:740\u201353.","journal-title":"N Engl J Med"},{"key":"3890_CR6","doi-asserted-by":"publisher","first-page":"956","DOI":"10.1200\/JCO.21.01693","volume":"40","author":"MM O\u2019Brien","year":"2022","unstructured":"O\u2019Brien MM, Ji L, Shah NN, Rheingold SR, Bhojwani D, Yuan CM, et al. Phase II trial of inotuzumab ozogamicin in children and adolescents with relapsed or refractory B-cell acute lymphoblastic leukemia: Children\u2019s Oncology Group protocol AALL1621. J Clin Oncol. 2022;40:956\u201367.","journal-title":"J Clin Oncol"},{"key":"3890_CR7","doi-asserted-by":"publisher","first-page":"1516","DOI":"10.1038\/s41375-022-01576-3","volume":"36","author":"E Pennesi","year":"2022","unstructured":"Pennesi E, Michels N, Brivio E, van der Velden VHJ, Jiang Y, Thano A, et al. Inotuzumab ozogamicin as single agent in pediatric patients with relapsed and refractory acute lymphoblastic leukemia: results from a phase II trial. Leukemia. 2022;36:1516\u201324.","journal-title":"Leukemia"},{"key":"3890_CR8","doi-asserted-by":"publisher","first-page":"1384","DOI":"10.1182\/bloodadvances.2023011514","volume":"8","author":"L Metheny","year":"2024","unstructured":"Metheny L, Sobecks R, Cho C, Fu P, Margevicius S, Wang J, et al. A multicenter study of posttransplant low-dose inotuzumab ozogamicin to prevent relapse of acute lymphoblastic leukemia. Blood Adv. 2024;8:1384\u201391.","journal-title":"Blood Adv"},{"key":"3890_CR9","doi-asserted-by":"publisher","first-page":"890","DOI":"10.1007\/s12185-022-03315-x","volume":"115","author":"Y Takahashi","year":"2022","unstructured":"Takahashi Y, Ishida H, Imamura T, Tamefusa K, Suenobu S, Usami I, et al. JACLS ALL-02 SR protocol reduced-intensity chemotherapy produces excellent outcomes in patients with low-risk childhood acute lymphoblastic leukemia. Int J Hematol. 2022;115:890\u20137.","journal-title":"Int J Hematol"},{"key":"3890_CR10","doi-asserted-by":"publisher","first-page":"2736","DOI":"10.1200\/JCO.2012.48.5680","volume":"31","author":"C Eckert","year":"2013","unstructured":"Eckert C, Henze G, Seeger K, Hagedorn N, Mann G, Panzer-Gr\u00fcmayer R, et al. Use of allogeneic hematopoietic stem-cell transplantation based on minimal residual disease response improves outcomes for children with relapsed acute lymphoblastic leukemia in the intermediate-risk group. J Clin Oncol. 2013;31:2736\u201342.","journal-title":"J Clin Oncol"},{"key":"3890_CR11","doi-asserted-by":"publisher","first-page":"274","DOI":"10.1111\/bjh.15919","volume":"186","author":"TM Horton","year":"2019","unstructured":"Horton TM, Whitlock JA, Lu X, O\u2019Brien MM, Borowitz MJ, Devidas M, et al. Bortezomib reinduction chemotherapy in high-risk ALL in first relapse: a report from the Children\u2019s Oncology Group. Br J Haematol. 2019;186:274\u201385.","journal-title":"Br J Haematol"},{"key":"3890_CR12","doi-asserted-by":"publisher","first-page":"417","DOI":"10.1182\/blood.2023022330","volume":"143","author":"E Jabbour","year":"2024","unstructured":"Jabbour E, Haddad FG, Short NJ, Senapati J, Jain N, Sasaki K, et al. Phase 2 study of inotuzumab ozogamicin for measurable residual disease in acute lymphoblastic leukemia in remission. Blood. 2024;143:417\u201321.","journal-title":"Blood"},{"key":"3890_CR13","doi-asserted-by":"publisher","first-page":"1582","DOI":"10.1182\/blood.2020007848","volume":"137","author":"E Brivio","year":"2021","unstructured":"Brivio E, Locatelli F, Lopez-Yurda M, Malone A, D\u00edaz-de-Heredia C, Bielorai B, et al. A phase 1 study of inotuzumab ozogamicin in pediatric relapsed\/refractory acute lymphoblastic leukemia (ITCC-059 study). Blood. 2021;137:1582\u201390.","journal-title":"Blood"}],"container-title":["International Journal of Hematology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s12185-024-03890-1.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1007\/s12185-024-03890-1\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s12185-024-03890-1.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,1,30]],"date-time":"2025-01-30T16:35:17Z","timestamp":1738254917000},"score":1,"resource":{"primary":{"URL":"https:\/\/link.springer.com\/10.1007\/s12185-024-03890-1"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,12,9]]},"references-count":13,"journal-issue":{"issue":"2","published-print":{"date-parts":[[2025,2]]}},"alternative-id":["3890"],"URL":"https:\/\/doi.org\/10.1007\/s12185-024-03890-1","relation":{},"ISSN":["0925-5710","1865-3774"],"issn-type":[{"value":"0925-5710","type":"print"},{"value":"1865-3774","type":"electronic"}],"subject":[],"published":{"date-parts":[[2024,12,9]]},"assertion":[{"value":"6 August 2024","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"23 November 2024","order":2,"name":"revised","label":"Revised","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"25 November 2024","order":3,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"9 December 2024","order":4,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"The authors have no potential conflicts of interest to declare.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Conflict of interest"}}]}}