{"doi":"10.1007/s12185-024-03890-1","title":"Inotuzumab ozogamicin for relapse prevention in a boy with Down syndrome and relapsed acute lymphoblastic leukemia","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>","journal":"International Journal of Hematology","year":2025,"id":633782,"datarank":0.26876392038420827,"base_score":1.791759469228055,"endowment":1.791759469228055,"self_citation_contribution":0.26876392038420827,"citation_network_contribution":0.0,"self_endowment_contribution":0.26876392038420827,"citer_contribution":0.0,"corpus_percentile":null,"corpus_rank":null,"citation_count":5,"citer_count":0,"citers_with_citation_signal":0,"citers_with_endowment":0,"datacite_reuse_total":0,"is_dataset":false,"is_dataset_confidence":null,"is_data_producer":false,"deposit_databanks":null,"is_oa":false,"file_count":0,"downloads":0,"has_version_chain":false,"published_date":null,"fair_score":null,"fair_percentile":null,"algorithm_id":"datarank_citation_only_1hop_v6","ranking_scope":"data_only","authors":[{"id":1643405,"name":"Hidemi Toyoda","orcid":"0000-0002-4727-6050","position":1,"is_corresponding":false},{"id":1643407,"name":"Ryo Hanaki","orcid":null,"position":2,"is_corresponding":false},{"id":1643408,"name":"Kaori Niwa","orcid":null,"position":3,"is_corresponding":false},{"id":1643411,"name":"Yosuke Okumura","orcid":null,"position":4,"is_corresponding":false},{"id":1643414,"name":"Mari Morimoto","orcid":null,"position":5,"is_corresponding":false},{"id":253605,"name":"Takahiro Ito","orcid":"0000-0002-1876-3052","position":6,"is_corresponding":false},{"id":1643418,"name":"Masahiro Hirayama","orcid":null,"position":7,"is_corresponding":false},{"id":1643404,"name":"Atsushi Kohso","orcid":null,"position":0,"is_corresponding":false}],"reference_count":0,"raw_metadata":{"has_enrichment":true,"resolved":true,"title":"Inotuzumab ozogamicin for relapse prevention in a boy with Down syndrome and relapsed acute lymphoblastic leukemia","abstract":"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.","is_dataset_classified":null,"base_score":1.791759469228055,"endowment":1.791759469228055,"datacite_reuse_total":0,"file_count":0,"downloads":0,"views":0,"has_version_chain":false,"is_dataset":false,"is_oa":false,"pmid":"39648271","pmcid":"PMC11782362","openalex_id":"https://openalex.org/W4405156229","authors":[],"funders":[],"total_grants":0,"fwci":2.0437,"citation_percentile":0.87720757,"influential_citations":0,"citation_trend":[{"year":2025,"count":4},{"year":2026,"count":1}],"oa_status":"hybrid","license":"cc-by","oa_locations":[{"url":"https://link.springer.com/content/pdf/10.1007/s12185-024-03890-1.pdf","host_type":"journal"},{"url":"https://link.springer.com/content/pdf/10.1007/s12185-024-03890-1.pdf","host_type":"publisher"},{"url":"https://doi.org/10.1007/s12185-024-03890-1","host_type":"journal"},{"url":"https://pubmed.ncbi.nlm.nih.gov/39648271","host_type":"repository"},{"url":"https://www.ncbi.nlm.nih.gov/pmc/articles/11782362","host_type":"repository"},{"url":"https://pmc.ncbi.nlm.nih.gov/articles/PMC11782362/pdf/12185_2024_Article_3890.pdf","host_type":"repository"},{"url":"https://europepmc.org/articles/PMC11782362","host_type":"Europe_PMC"},{"url":"https://europepmc.org/articles/PMC11782362?pdf=render","host_type":"Europe_PMC"}],"fields_of_study":["Acute Lymphoblastic Leukemia research","Chronic Myeloid Leukemia Treatments","Lymphoma Diagnosis and Treatment","Humans","Inotuzumab Ozogamicin","Down Syndrome","Male","Secondary Prevention","Precursor Cell Lymphoblastic Leukemia-Lymphoma","Recurrence","Antibodies, Monoclonal, Humanized","Antineoplastic Agents, Immunological","Hematopoietic Stem Cell Transplantation","Child"],"mesh_terms":["Antineoplastic Agents, Immunological","Inotuzumab Ozogamicin","Child","Down Syndrome","Humans","Male","Recurrence","Remission Induction","Precursor B-Cell Lymphoblastic Leukemia-Lymphoma","Hematopoietic Stem Cell Transplantation","Precursor Cell Lymphoblastic Leukemia-Lymphoma","Secondary Prevention","Antibodies, Monoclonal, Humanized"],"keywords":["Calicheamicin","Medicine","Refractory (planetary science)","Internal medicine","Hematology","Lymphoblastic Leukemia","Population","Blinatumomab","Gemtuzumab ozogamicin","CD22","Complete remission","Salvage therapy","Oncology","Leukemia","Chemotherapy","CD34","Lymphoma","Stem cell","CD33","CD20","Relapse","Maintenance Chemotherapy","B-cell Precursor Acute Lymphoblastic Leukemia","Inotuzumab Ozogamicin"],"sdg_mappings":[{"sdg_number":0,"sdg_label":"Good health and well-being"}],"linked_datasets":[],"clinical_trials":[],"software_tools":[],"database_accessions":[],"source":"live","citation_network_status":"fetched"},"created_at":"2026-08-06T12:39:47.406204Z","pmid":null,"pmcid":null,"fwci":null,"citation_percentile":null,"influential_citations":0,"oa_status":null,"license":null,"views":0,"total_file_size_bytes":0,"version_count":0,"fair_f":null,"fair_a":null,"fair_i":null,"fair_r":null,"fair_zscore":null,"fair_rationale":null,"fair_model":null,"fair_agent_version":null,"fair_fulltext_source":null,"fair_has_llm":null,"fair_computed_at":null,"clinical_trials":[],"software_tools":[],"db_accessions":[],"linked_datasets":[],"topics":[]}