{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,1]],"date-time":"2026-08-01T11:32:30Z","timestamp":1785583950026,"version":"3.56.0"},"reference-count":19,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2025,6,18]],"date-time":"2025-06-18T00:00:00Z","timestamp":1750204800000},"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. Nephrol."],"abstract":"<jats:p>Catastrophic antiphospholipid syndrome (CAPS) leads to organ dysfunction due to thrombotic microangiopathy (TMA). Complement may play a role in CAPS, and its blockade could prevent antiphospholipid syndrome (APS) complications after kidney transplantation (KT). Here, we report a case of APS recurrence after KT in a 38-year-old woman with early acute cortical kidney allograft necrosis despite preventive eculizumab treatment, probably because of insufficient complement blockade. The patient had recurrent but controlled CAPS for years with renal dysfunction, leading to preemptive KT. Anticoagulation and eculizumab were administered to prevent thrombosis and TMA after KT. She developed acute kidney injury (AKI) with incomplete biological TMA. Imaging revealed cortical necrosis in the renal allograft. In the absence of donor-specific anti-HLA antibodies, we concluded a relapse. Additional doses of eculizumab and plasma exchange allowed the normalization of biological tests and improvement of kidney allograft function. A retrospective complement analysis showed an incomplete blockade at the time of AKI. One year after KT, the renal allograft function was impaired. This suggests that inadequate complement blockade leads to a relapse of APS in the renal allograft with cortical necrosis and dysfunction. Our case highlights the importance of monitoring complement activity and adjusting the dose of eculizumab or ravulizumab.<\/jats:p>","DOI":"10.3389\/fneph.2025.1572641","type":"journal-article","created":{"date-parts":[[2025,6,18]],"date-time":"2025-06-18T04:16:33Z","timestamp":1750220193000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":3,"title":["Case Report: Failure of eculizumab to block complement to prevent relapse of anti-phospholipid syndrome in kidney transplant recipient"],"prefix":"10.3389","volume":"5","author":[{"given":"Thibault","family":"Laban","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Fred\u00e9ric","family":"Pigneur","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Constance","family":"Guillaud","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marie Agn\u00e8s","family":"Dragon Durey","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Houcine","family":"Hamidi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Caroline","family":"Pilon","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marc","family":"Michel","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nizar","family":"Joher","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Philippe","family":"Grimbert","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hamza","family":"Sakhi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Antoine","family":"Morel","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marie","family":"Matignon","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1965","published-online":{"date-parts":[[2025,6,18]]},"reference":[{"key":"B1","doi-asserted-by":"publisher","DOI":"10.3390\/antib13010021","article-title":"Diagnosis and management of catastrophic antiphospholipid syndrome and the potential impact of the 2023 ACR\/EULAR antiphospholipid syndrome classification criteria","volume":"13","author":"Jacobs","year":"2024","journal-title":"Antibodies"},{"key":"B2","doi-asserted-by":"publisher","DOI":"10.1111\/ajt.12540","article-title":"Eculizumab prevents recurrent antiphospholipid antibody syndrome and enables successful renal transplantation","volume":"14","author":"Lonze","year":"2014","journal-title":"Am J Transplantation"},{"key":"B3","doi-asserted-by":"publisher","DOI":"10.1182\/blood.2020007499","article-title":"Patients with refractory catastrophic antiphospholipid syndrome respond inconsistently to eculizumab","volume":"136","author":"Yelnik","year":"2020","journal-title":"Blood"},{"key":"B4","doi-asserted-by":"publisher","DOI":"10.1136\/annrheumdis-2019-215213","article-title":"EULAR recommendations for the management of antiphospholipid syndrome in adults","volume":"78","author":"Tektonidou","year":"2019","journal-title":"Ann Rheum Dis"},{"key":"B5","doi-asserted-by":"publisher","DOI":"10.1016\/j.cca.2023.117750","article-title":"Usefulness and analytical performances of complement multiplex assay for measuring complement biomarkers in plasma","volume":"554","author":"Meuleman","year":"2024","journal-title":"Clin Chim Acta"},{"key":"B6","doi-asserted-by":"publisher","DOI":"10.3389\/fimmu.2018.01130","article-title":"Endothelial dysfunction in kidney transplantation","volume":"9","author":"Cardinal","year":"2018","journal-title":"Front Immunol"},{"key":"B7","doi-asserted-by":"publisher","DOI":"10.1016\/j.kint.2021.11.028","article-title":"Thirty years of the International Banff Classification for Allograft Pathology: the past, present, and future of kidney transplant diagnostics","volume":"101","author":"Loupy","year":"2022","journal-title":"Kidney Int"},{"key":"B8","doi-asserted-by":"publisher","DOI":"10.1097\/MD.0000000000025958","article-title":"Early critical cortical infarction by anti-angiotensin II type 1 receptor antibody: A case report and literature review","volume":"100","author":"Lim","year":"2021","journal-title":"Med (Baltimore)"},{"key":"B9","doi-asserted-by":"publisher","first-page":"692","DOI":"10.1681\/ASN.2018080868","article-title":"Early acute microvascular kidney transplant rejection in the absence of anti-HLA antibodies is associated with preformed igG antibodies against diverse glomerular endothelial cell antigens","volume":"30","author":"Delville","year":"2019","journal-title":"J Am Soc Nephrol"},{"key":"B10","doi-asserted-by":"publisher","DOI":"10.1097\/TP.0000000000001231","article-title":"Anti-angiotensin II type 1 receptor and anti-endothelial cell antibodies: A cross-sectional analysis of pathological findings in allograft biopsies","volume":"101","author":"Philogene","year":"2017","journal-title":"Transplantation"},{"key":"B11","doi-asserted-by":"publisher","DOI":"10.1093\/ndt\/gfv375","article-title":"The clinicopathological relevance of pretransplant anti-angiotensin II type 1 receptor antibodies in renal transplantation","volume":"32","author":"Lee","year":"2017","journal-title":"Nephrol Dial Transplant"},{"key":"B12","doi-asserted-by":"publisher","DOI":"10.1111\/ajt.15074","article-title":"Histological picture of antibody-mediated rejection without donor-specific anti-HLA antibodies: Clinical presentation and implications for outcome","volume":"19","author":"Senev","year":"2019","journal-title":"Am J Transplant"},{"key":"B13","doi-asserted-by":"publisher","DOI":"10.1097\/00007890-200004150-00023","article-title":"Frequency, potential risk and therapeutic intervention in end-stage renal disease patients with antiphospholipid antibody syndrome: a multicenter study","volume":"69","author":"Vaidya","year":"2000","journal-title":"Transplantation"},{"key":"B14","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2005-03-1319","article-title":"Thrombus formation induced by antibodies to beta2-glycoprotein I is complement dependent and requires a priming factor","volume":"106","author":"Fischetti","year":"2005","journal-title":"Blood"},{"key":"B15","doi-asserted-by":"publisher","DOI":"10.1111\/ajt.12319","article-title":"Eculizumab improves posttransplant thrombotic microangiopathy due to antiphospholipid syndrome recurrence but fails to prevent chronic vascular changes","volume":"13","author":"Canaud","year":"2013","journal-title":"Am J Transplantation"},{"key":"B16","article-title":"Complement activity and complement regulatory gene mutations are associated with thrombosis in APS and CAPS","volume":"135","author":"Chaturvedi","year":"2020","journal-title":"Blood J Am Soc Hematol"},{"key":"B17","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMra1705454","article-title":"Diagnosis and management of the antiphospholipid syndrome","volume":"378","author":"Garcia","year":"2018","journal-title":"N Engl J Med"},{"key":"B18","doi-asserted-by":"publisher","DOI":"10.1182\/blood.2020009280","article-title":"Eculizumab discontinuation in children and adults with atypical hemolytic-uremic syndrome: a prospective multicenter study","volume":"137","author":"Fakhouri","year":"2021","journal-title":"Blood"},{"key":"B19","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2018-09-876136","article-title":"Ravulizumab (ALXN1210) vs eculizumab in adult patients with PNH naive to complement inhibitors: the 301 study","volume":"133","author":"Lee","year":"2019","journal-title":"Blood"}],"container-title":["Frontiers in Nephrology"],"original-title":[],"link":[{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fneph.2025.1572641\/full","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,6,18]],"date-time":"2025-06-18T04:16:34Z","timestamp":1750220194000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fneph.2025.1572641\/full"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,6,18]]},"references-count":19,"alternative-id":["10.3389\/fneph.2025.1572641"],"URL":"https:\/\/doi.org\/10.3389\/fneph.2025.1572641","relation":{},"ISSN":["2813-0626"],"issn-type":[{"value":"2813-0626","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,6,18]]},"article-number":"1572641"}}