{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,4,21]],"date-time":"2026-04-21T02:12:41Z","timestamp":1776737561943,"version":"3.51.2"},"reference-count":63,"publisher":"MDPI AG","issue":"6","license":[{"start":{"date-parts":[[2024,3,11]],"date-time":"2024-03-11T00:00:00Z","timestamp":1710115200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["JCM"],"abstract":"<jats:p>Thrombotic microangiopathy (TMA) has been observed in some patients receiving interferon beta (IFN\u03b2) therapy for relapsing-remitting multiple sclerosis, but little is known about its clinical features and outcomes. We searched the literature to identify cases with IFN\u03b2-related TMA and assessed their pattern of organ involvement, the presence of prodromal manifestations, the treatments used, and the outcomes. Thirty-five articles met the inclusion criteria, and data of 67 patients were collected. The median duration of IFN\u03b2 therapy before the diagnosis of TMA was 8 years, and 56\/67 (84%) presented with acute kidney injury (AKI), of which 33 required acute dialysis. All but three patients had manifestations during the four weeks before TMA onset, including flu-like symptoms, headache, and worsening blood pressure control. In only two patients, ADAMTS13 activity was reduced, while 27% had low C3 levels. However, none showed causative genetic mutations associated with development of atypical hemolytic uremic syndrome. All patients discontinued IFN\u03b2, 34 (55%) also received plasma exchange, and 12 (18%) received eculizumab. Complete renal recovery was achieved by 20 patients (30%), while 13 (20%) developed end-stage renal disease. Among those with AKI requiring dialysis, eculizumab therapy was associated with a significantly reduced risk of ESRD compared with plasma exchange. Therefore, TMA with features of aHUS mainly occurs after prolonged treatment with IFN\u03b2 and is preceded by prodromes, which may lead to an early diagnosis before life-threatening complications occur. Eculizumab appears beneficial in cases with severe kidney involvement, which supports a role of the complement system in the pathogenesis of these forms.<\/jats:p>","DOI":"10.3390\/jcm13061598","type":"journal-article","created":{"date-parts":[[2024,3,11]],"date-time":"2024-03-11T04:51:12Z","timestamp":1710132672000},"page":"1598","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":4,"title":["Thrombotic Microangiopathy as a Life-Threatening Complication of Long-Term Interferon Beta Therapy for Multiple Sclerosis: Clinical Phenotype and Response to Treatment\u2014A Literature Review"],"prefix":"10.3390","volume":"13","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-9949-3543","authenticated-orcid":false,"given":"Marco","family":"Allinovi","sequence":"first","affiliation":[{"name":"Nephrology, Dialysis and Transplantation Unit, Careggi University Hospital, 50134 Florence, Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Tommaso","family":"Mazzierli","sequence":"additional","affiliation":[{"name":"Nephrology, Dialysis and Transplantation Unit, Careggi University Hospital, 50134 Florence, Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Selene","family":"Laudicina","sequence":"additional","affiliation":[{"name":"Nephrology, Dialysis and Transplantation Unit, Careggi University Hospital, 50134 Florence, Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Luisa","family":"Past\u00f2","sequence":"additional","affiliation":[{"name":"NEUROFARBA Department, University of Florence, 50139 Florence, Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Emilio","family":"Portaccio","sequence":"additional","affiliation":[{"name":"NEUROFARBA Department, University of Florence, 50139 Florence, Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Maria Pia","family":"Amato","sequence":"additional","affiliation":[{"name":"NEUROFARBA Department, University of Florence, 50139 Florence, Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1578-4402","authenticated-orcid":false,"given":"Giorgio","family":"Trivioli","sequence":"additional","affiliation":[{"name":"Department of Nephrology, Cambridge University Hospital NHS Foundation Trust, Cambridge CB2 0QQ, UK"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1968","published-online":{"date-parts":[[2024,3,11]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"771","DOI":"10.1080\/14737175.2020.1721282","article-title":"Immunosuppression in Relapsing Remitting Multiple Sclerosis: Moving towards Personalized Treatment","volume":"20","author":"Patti","year":"2020","journal-title":"Expert Rev. 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