{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,12]],"date-time":"2026-10-12T14:03:10Z","timestamp":1791813790486,"version":"4.3.4"},"reference-count":13,"publisher":"Wiley","license":[{"start":{"date-parts":[[2026,10,12]],"date-time":"2026-10-12T00:00:00Z","timestamp":1791763200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"},{"start":{"date-parts":[[2026,10,12]],"date-time":"2026-10-12T00:00:00Z","timestamp":1791763200000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Pediatric Blood &amp;amp; Cancer"],"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>Cold antibody\u2010mediated autoimmune hemolytic anemia, or cold agglutinin syndrome (CAS), in pediatrics is typically transient, although it can often be severe and require transfusion support. Recurrence in children is rare, but those with underlying immune dysfunction can have a refractory course with limited options available. We report a 4\u2010year\u2010old girl with Evans syndrome and life\u2010threatening CAS refractory to transfusions, plasmapheresis, steroids, and immunosuppression as well as a 12\u2010month\u2010old boy status post liver transplant with CAS refractory to steroids, rituximab, and tacrolimus cessation with transition to sirolimus, both of whom received sutimlimab, a C1s inhibitor, with rapid improvement in hemolysis, transfusion independence, and a sustained remission. To our knowledge, these are the first reported uses of sutimlimab in the pediatric population.<\/jats:p>","DOI":"10.1002\/1545-5017.70751","type":"journal-article","created":{"date-parts":[[2026,10,12]],"date-time":"2026-10-12T13:41:15Z","timestamp":1791812475000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Successful Use of Sutimlimab in two Pediatric Patients With Refractory Cold Antibody\u2010Mediated Autoimmune Hemolytic Anemia"],"prefix":"10.1002","author":[{"ORCID":"https:\/\/orcid.org\/0009-0009-6279-4743","authenticated-orcid":false,"given":"Sibgha","family":"Zaheer","sequence":"first","affiliation":[{"name":"Children's Hospital Los Angeles  Los Angeles California USA"},{"name":"University of Southern California Keck School of Medicine  Los Angeles California USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rachel","family":"Friedmann","sequence":"additional","affiliation":[{"name":"Children's Hospital Los Angeles  Los Angeles California USA"},{"name":"University of Southern California Keck School of Medicine  Los Angeles California USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0000-3587-6414","authenticated-orcid":false,"given":"Kimberly","family":"Cheng","sequence":"additional","affiliation":[{"name":"Children's Hospital Los Angeles  Los Angeles California USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6013-1254","authenticated-orcid":false,"given":"Guy","family":"Young","sequence":"additional","affiliation":[{"name":"Children's Hospital Los Angeles  Los Angeles California USA"},{"name":"University of Southern California Keck School of Medicine  Los Angeles California USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3070-1775","authenticated-orcid":false,"given":"Christopher","family":"Denton","sequence":"additional","affiliation":[{"name":"Seattle Children's Hospital  Seattle Washington USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2875-6948","authenticated-orcid":false,"given":"Taylor","family":"Kim","sequence":"additional","affiliation":[{"name":"Children's Hospital Los Angeles  Los Angeles California USA"},{"name":"University of Southern California Keck School of Medicine  Los Angeles California USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,10,12]]},"reference":[{"issue":"2","key":"e_1_2_7_2_1","doi-asserted-by":"crossref","first-page":"531","DOI":"10.1182\/bloodadvances.2025018452","article-title":"Cold Clues in Pediatric Autoimmune Hemolytic Anemia","volume":"10","author":"Jacobs J. W.","year":"2026","journal-title":"Blood Advances"},{"issue":"14","key":"e_1_2_7_3_1","doi-asserted-by":"crossref","first-page":"1323","DOI":"10.1056\/NEJMoa2027760","article-title":"Sutimlimab in Cold Agglutinin Disease","volume":"384","author":"R\u00f6th A.","year":"2021","journal-title":"New England Journal of Medicine"},{"issue":"9","key":"e_1_2_7_4_1","doi-asserted-by":"crossref","first-page":"980","DOI":"10.1182\/blood.2021014955","article-title":"Sutimlimab in Patients With Cold Agglutinin Disease: Results of the Randomized Placebo\u2010Controlled Phase 3 CADENZA Trial","volume":"140","author":"R\u00f6th A.","year":"2022","journal-title":"Blood"},{"key":"e_1_2_7_5_1","doi-asserted-by":"crossref","first-page":"590","DOI":"10.3389\/fimmu.2020.00590","article-title":"New Insights in the Pathogenesis and Therapy of Cold Agglutinin\u2010Mediated Autoimmune Hemolytic Anemia","volume":"11","author":"Berentsen S.","year":"2020","journal-title":"Frontiers in Immunology"},{"key":"e_1_2_7_6_1","volume-title":"Enjaymo [Package Insert]","year":"2024"},{"issue":"2","key":"e_1_2_7_7_1","doi-asserted-by":"crossref","first-page":"143","DOI":"10.1124\/jpet.122.001511","article-title":"Sutimlimab Pharmacokinetics and Pharmacodynamics in Patients With Cold Agglutinin Disease","volume":"386","author":"Frank T.","year":"2023","journal-title":"Journal of Pharmacology and Experimental Therapeutics"},{"issue":"4","key":"e_1_2_7_8_1","doi-asserted-by":"crossref","first-page":"655","DOI":"10.1002\/cpt.1111","article-title":"A Randomized, First\u2010in\u2010Human, Healthy Volunteer Trial of sutimlimab, a Humanized Antibody for the Specific Inhibition of the Classical Complement Pathway","volume":"104","author":"Bartko J.","year":"2018","journal-title":"Clinical Pharmacology & Therapeutics"},{"issue":"8","key":"e_1_2_7_9_1","doi-asserted-by":"crossref","first-page":"e617","DOI":"10.1016\/S2352-3026(24)00144-3","article-title":"Diagnosis and Management of Evans Syndrome in Adults: First Consensus Recommendations","volume":"11","author":"Fattizzo B.","year":"2024","journal-title":"Lancet Haematology"},{"key":"e_1_2_7_10_1","volume-title":"Soliris [Package Insert]","author":"Alexion","year":"2007"},{"issue":"19","key":"e_1_2_7_11_1","doi-asserted-by":"crossref","first-page":"2543","DOI":"10.1182\/bloodadvances.2018024190","article-title":"Eculizumab in Cold Agglutinin Disease (DECADE): An Open\u2010Label, Prospective, Bicentric, Nonrandomized Phase 2 Trial","volume":"2","author":"R\u00f6th A.","year":"2018","journal-title":"Blood Advances"},{"issue":"1","key":"e_1_2_7_12_1","doi-asserted-by":"crossref","first-page":"90","DOI":"10.1182\/hematology.2022000369","article-title":"Cold AIHA and the Best Treatment Strategies","volume":"2022","author":"Despotovic J. M.","year":"2022","journal-title":"Hematology: The American Society of Hematology Education Program"},{"issue":"10","key":"e_1_2_7_13_1","doi-asserted-by":"crossref","first-page":"1295","DOI":"10.1182\/blood.2019003809","article-title":"How I Treat Cold Agglutinin Disease","volume":"137","author":"Berentsen S.","year":"2021","journal-title":"Blood"},{"issue":"15","key":"e_1_2_7_14_1","doi-asserted-by":"crossref","first-page":"1407","DOI":"10.1056\/NEJMra2033982","article-title":"Autoimmune Hemolytic Anemias","volume":"385","author":"Berentsen S.","year":"2021","journal-title":"New England Journal of Medicine"}],"container-title":["Pediatric Blood &amp; Cancer"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/1545-5017.70751","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1002\/1545-5017.70751","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/1545-5017.70751","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,10,12]],"date-time":"2026-10-12T13:41:17Z","timestamp":1791812477000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/1545-5017.70751"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,10,12]]},"references-count":13,"alternative-id":["10.1002\/1545-5017.70751"],"URL":"https:\/\/doi.org\/10.1002\/1545-5017.70751","archive":["Portico"],"relation":{},"ISSN":["1545-5009","1545-5017"],"issn-type":[{"value":"1545-5009","type":"print"},{"value":"1545-5017","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,10,12]]},"assertion":[{"value":"2026-02-16","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-09-29","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-10-12","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"e70751"}}