{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,4]],"date-time":"2026-05-04T02:25:51Z","timestamp":1777861551381,"version":"3.51.4"},"reference-count":21,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2026,4,30]],"date-time":"2026-04-30T00:00:00Z","timestamp":1777507200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100013114","name":"Yunnan Key Research and Development Program","doi-asserted-by":"publisher","award":["202402AC100005"],"award-info":[{"award-number":["202402AC100005"]}],"id":[{"id":"10.13039\/501100013114","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"publisher","award":["82360036, 82060031"],"award-info":[{"award-number":["82360036, 82060031"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Immunol."],"abstract":"\n Background<\/jats:title>\n Low-dose immune tolerance induction (ITI) combined with standard-dose rituximab-based immunosuppression (LD-ITI + RTX-IS) is an effective strategy for factor VIII (FVIII) inhibitor eradication in severe hemophilia A with poor-risk prognostic factors for ITI, but optimal rituximab dosing remains undefined. Prolonged rituximab exposure increases adverse effects and financial burden.<\/jats:p>\n <\/jats:sec>\n \n Aim<\/jats:title>\n To describe the outcomes and tolerability of LD-ITI combined with single-dose rituximab (LD-ITI + SD-RTX-IS) for FVIII inhibitor eradication in severe hemophilia A.<\/jats:p>\n <\/jats:sec>\n \n Methods<\/jats:title>\n A single-center retrospective cohort study was conducted in patients with severe hemophilia A and inhibitors who received LD-ITI + SD-RTX-IS. Treatment comprised FVIII (50 IU\/kg every other day), rituximab (375 mg\/m\u00b2, single dose), and prednisone (1 mg\/kg daily, tapering). Success was defined as inhibitor titer &lt;0.6 BU\/mL and FVIII recovery \u226566%.<\/jats:p>\n <\/jats:sec>\n \n Results<\/jats:title>\n A total of 16 patients (median age 17 years) were enrolled, comprising 8 adults and 8 children. Eleven of 16 patients (68.8%) achieved successful inhibitor eradication with a median time of 9.3 months (IQR: 2.8\u201315.2; range: 1.8\u201318.6); median FVIII recovery was 82%. The monthly bleeding rate decreased by 84.2% from pre-ITI (median: 4 episodes) to during ITI (median: 0.66 episodes), with a corresponding reduction in monthly joint bleeds from 3 to 0.46 episodes. At 24-month follow-up, no recurrence was documented. Adverse events were predominantly mild (rituximab-related infusion reactions, n=2; fever\/pneumonia, n=4).<\/jats:p>\n <\/jats:sec>\n \n Conclusions<\/jats:title>\n In this cohort, LD-ITI + SD-RTX-IS was associated with an inhibitor eradication rate consistent with prior LD-ITI + RTX-IS reports, using less rituximab. These preliminary findings warrant further prospective evaluation.<\/jats:p>\n <\/jats:sec>","DOI":"10.3389\/fimmu.2026.1829007","type":"journal-article","created":{"date-parts":[[2026,4,30]],"date-time":"2026-04-30T04:25:47Z","timestamp":1777523147000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":0,"title":["Outcomes of low-dose immune tolerance induction with single-dose rituximab in severe hemophilia A: a single-center retrospective experience"],"prefix":"10.3389","volume":"17","author":[{"given":"Shaoyu","family":"Yin","sequence":"first","affiliation":[{"name":"Department of Hematology, the Second Affiliated Hospital of Kunming Medical University","place":["Kunming, China"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hongli","family":"Mu","sequence":"additional","affiliation":[{"name":"Department of Hematology, the Second Affiliated Hospital of Kunming Medical University","place":["Kunming, 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