{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,7,30]],"date-time":"2025-07-30T16:01:27Z","timestamp":1753891287653,"version":"3.41.2"},"reference-count":25,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2023,8,29]],"date-time":"2023-08-29T00:00:00Z","timestamp":1693267200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Med."],"abstract":"<jats:p>Type B insulin resistance (TBIR) is a rare, often fulminant form of insulin resistance caused by autoantibodies against the insulin receptor. If left untreated, its mortality is high. Various immunosuppressive regimens have shown efficacy, but treatment effects are variable and time-delayed, and drug-induced complications may arise. We report a patient with TBIR arising as a complication of Wiskott\u2013Aldrich syndrome. Stable remission of TBIR was achieved through allogeneic peripheral blood stem cell transplantation (PBSCT) over a follow-up period of more than 1.5 years. We thus demonstrate that PBSCT can be considered a treatment option in TBIR where conventional immunosuppressive therapy is ineffective or contraindicated.<\/jats:p>","DOI":"10.3389\/fmed.2023.1200037","type":"journal-article","created":{"date-parts":[[2023,8,29]],"date-time":"2023-08-29T22:49:32Z","timestamp":1693349372000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":1,"title":["Case report: Allogeneic stem cell transplantation for type B insulin resistance"],"prefix":"10.3389","volume":"10","author":[{"given":"Thomas","family":"Ebert","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Gerhard","family":"Behre","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Lorenz","family":"Weidhase","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Vladan","family":"Vucinic","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Cornelia","family":"Gewert","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Robert K.","family":"Semple","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Michael","family":"Stumvoll","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Anke","family":"T\u00f6njes","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1965","published-online":{"date-parts":[[2023,8,29]]},"reference":[{"key":"B1","doi-asserted-by":"publisher","first-page":"739","DOI":"10.1056\/NEJM197604012941401","article-title":"The syndromes of insulin resistance and acanthosis Nigricans","volume":"294","author":"Kahn","year":"1976","journal-title":"N Engl J Med."},{"key":"B2","doi-asserted-by":"publisher","first-page":"4225","DOI":"10.1172\/JCI142245","article-title":"Severe insulin resistance syndromes","volume":"131","author":"Angelidi","year":"2021","journal-title":"J Clin Invest"},{"key":"B3","doi-asserted-by":"publisher","first-page":"87","DOI":"10.1097\/00005792-200203000-00001","article-title":"Clinical course of the syndrome of autoantibodies to the insulin receptor (type B Insulin Resistance): a 28-year perspective","volume":"81","author":"Arioglu","year":"2002","journal-title":"Medicine."},{"key":"B4","doi-asserted-by":"publisher","first-page":"1094","DOI":"10.1172\/JCI108861","article-title":"Effects of Autoantibodies to the Insulin Receptor on Isolated Adipocytes: Stud Insulin Binding Insulin Action","volume":"60","author":"Kahn","year":"1977","journal-title":"J Clin Invest"},{"key":"B5","doi-asserted-by":"publisher","first-page":"1072","DOI":"10.1172\/JCI111032","article-title":"Clinical disorders associated with autoantibodies to the insulin receptor. Simulation by passive transfer of immunoglobulins to rats","volume":"72","author":"Dons","year":"1983","journal-title":"J Clin Invest."},{"key":"B6","doi-asserted-by":"publisher","first-page":"318","DOI":"10.1097\/MED.0000000000000263","article-title":"Type B insulin resistance syndrome","volume":"23","author":"Willard","year":"2016","journal-title":"Curr Opin Endocrinol Diabetes Obes."},{"key":"B7","doi-asserted-by":"publisher","first-page":"1719","DOI":"10.1210\/jc.2014-3552","article-title":"Successful treatment of type B insulin resistance with rituximab","volume":"100","author":"Manikas","year":"2015","journal-title":"J Clin Endocrinol Metab."},{"key":"B8","doi-asserted-by":"publisher","first-page":"835","DOI":"10.1038\/ncpendmet0693","article-title":"patient with type B insulin resistance syndrome, responsive to immune therapy","volume":"3","author":"Page","year":"2007","journal-title":"Nat Clin Pract Endocrinol Metab."},{"key":"B9","doi-asserted-by":"publisher","first-page":"3641","DOI":"10.1210\/jc.2010-0167","article-title":"Treatment of type B insulin resistance: a novel approach to reduce insulin receptor autoantibodies","volume":"95","author":"Malek","year":"2010","journal-title":"J Clin Endocrinol Metab."},{"key":"B10","doi-asserted-by":"publisher","first-page":"1217","DOI":"10.2337\/diacare.21.8.1217","article-title":"Successful treatment with plasmapheresis, cyclophosphamide, and cyclosporin A in type B syndrome of insulin resistance: case report","volume":"21","author":"Eriksson","year":"1998","journal-title":"Diabetes Care."},{"key":"B11","doi-asserted-by":"publisher","first-page":"310","DOI":"10.1056\/NEJM200401153500324","article-title":"Rituximab therapy for the type B syndrome of severe insulin resistance","volume":"350","author":"Coll","year":"2004","journal-title":"N Engl J Med."},{"key":"B12","doi-asserted-by":"publisher","first-page":"2353","DOI":"10.2337\/dc18-0884","article-title":"Combined immunosuppressive therapy induces remission in patients with severe type B insulin resistance: a prospective cohort study","volume":"41","author":"Klubo-Gwiezdzinska","year":"2018","journal-title":"Diabetes Care."},{"key":"B13","doi-asserted-by":"publisher","first-page":"13","DOI":"10.1007\/s10875-017-0453-z","article-title":"Clinical Manifestations and Pathophysiological Mechanisms of the Wiskott-Aldrich Syndrome","volume":"38","author":"Candotti","year":"2018","journal-title":"J Clin Immunol."},{"key":"B14","doi-asserted-by":"publisher","first-page":"814","DOI":"10.1111\/j.1464-5491.2005.01529.x","article-title":"Successful treatment of Type B insulin resistance in a patient with otherwise quiescent systemic lupus erythematosus","volume":"22","author":"Coll","year":"2005","journal-title":"Diabet Med."},{"key":"B15","doi-asserted-by":"publisher","first-page":"2066","DOI":"10.1182\/blood.2021014687","article-title":"Hematopoietic stem cell transplantation for Wiskott-Aldrich syndrome: an EBMT inborn errors working party analysis","volume":"139","author":"Albert","year":"2022","journal-title":"Blood."},{"key":"B16","doi-asserted-by":"publisher","first-page":"3390","DOI":"10.1182\/blood.V97.11.3390","article-title":"Hematopoietic cell transplantation in older patients with hematologic malignancies: replacing high-dose cytotoxic therapy with graft-vs","volume":"97","author":"McSweeney","year":"2001","journal-title":"Blood."},{"key":"B17","doi-asserted-by":"publisher","first-page":"1143","DOI":"10.1016\/j.bbmt.2009.06.019","article-title":"Guidelines for preventing infectious complications among hematopoietic cell transplantation recipients: a global perspective","volume":"15","author":"Tomblyn","year":"2009","journal-title":"Biol Blood Marrow Transplant."},{"key":"B18","doi-asserted-by":"publisher","first-page":"4010","DOI":"10.1182\/blood-2003-05-1592","article-title":"Mutations of the Wiskott-Aldrich syndrome protein (WASP): hotspots, effect on transcription, and translation and phenotype\/genotype correlation","volume":"104","author":"Jin","year":"2004","journal-title":"Blood."},{"key":"B19","doi-asserted-by":"publisher","first-page":"1995","DOI":"10.1093\/hmg\/4.10.1995","article-title":"Scanning of the Wiskott-Aldrich syndrome (WAS) gene: identification of 18 novel alterations including a possible mutation hotspot at Arg86 resulting in thrombocytopenia, a mild WAS phenotype","volume":"4","author":"Kwan","year":"1995","journal-title":"Hum Mol Genet."},{"key":"B20","doi-asserted-by":"publisher","first-page":"977","DOI":"10.2337\/dc07-2194","article-title":"Plasma adiponectin as a marker of insulin receptor dysfunction: clinical utility in severe insulin resistance","volume":"31","author":"Semple","year":"2008","journal-title":"Diabetes Care."},{"key":"B21","doi-asserted-by":"publisher","first-page":"2912","DOI":"10.1182\/blood-2005-05-2004","article-title":"Hematopoietic cell transplantation (HCT)-specific comorbidity index: a new tool for risk assessment before allogeneic HCT","volume":"106","author":"Sorror","year":"2005","journal-title":"Blood."},{"key":"B22","doi-asserted-by":"publisher","first-page":"1525","DOI":"10.1038\/s41409-019-0516-2","article-title":"Indications for haematopoietic stem cell transplantation for haematological diseases, solid tumours and immune disorders: current practice in Europe, 2019","volume":"54","author":"Duarte","year":"2019","journal-title":"Bone Marrow Transplant."},{"key":"B23","doi-asserted-by":"publisher","first-page":"470","DOI":"10.1126\/science.adg7489","article-title":"Resetting tolerance in autoimmune disease","volume":"380","author":"Muraro","year":"2023","journal-title":"Science."},{"key":"B24","doi-asserted-by":"publisher","first-page":"215","DOI":"10.1146\/annurev-med-070119-115617","article-title":"Hematopoietic stem cell transplantation for autoimmune disease","volume":"72","author":"Alexander","year":"2021","journal-title":"Annu Rev Med."},{"key":"B25","doi-asserted-by":"publisher","first-page":"1758","DOI":"10.1056\/NEJM200106073442304","article-title":"Treatment of the immune dysregulation, polyendocrinopathy, enteropathy, X-linked syndrome (IPEX) by allogeneic bone marrow transplantation","volume":"344","author":"Baud","year":"2001","journal-title":"N Engl J Med."}],"container-title":["Frontiers in Medicine"],"original-title":[],"link":[{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fmed.2023.1200037\/full","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,8,29]],"date-time":"2023-08-29T22:49:35Z","timestamp":1693349375000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fmed.2023.1200037\/full"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,8,29]]},"references-count":25,"alternative-id":["10.3389\/fmed.2023.1200037"],"URL":"https:\/\/doi.org\/10.3389\/fmed.2023.1200037","relation":{},"ISSN":["2296-858X"],"issn-type":[{"type":"electronic","value":"2296-858X"}],"subject":[],"published":{"date-parts":[[2023,8,29]]},"article-number":"1200037"}}