{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,18]],"date-time":"2026-05-18T09:07:04Z","timestamp":1779095224672,"version":"3.51.4"},"reference-count":14,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2026,5,18]],"date-time":"2026-05-18T00:00:00Z","timestamp":1779062400000},"content-version":"vor","delay-in-days":137,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2026,1,1]],"date-time":"2026-01-01T00:00:00Z","timestamp":1767225600000},"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":["Case Reports in Critical Care"],"published-print":{"date-parts":[[2026,1]]},"abstract":"<jats:p>Autoimmune polyendocrine syndrome Type 1, also known as autoimmune polyendocrinopathy\u2013candidiasis\u2013ectodermal dystrophy (APS\u20101\/APECED), is a rare monogenic autoimmune disorder with increasing recognition of neurologic manifestations. We report a critically ill adolescent female with APS\u20101 and a prior history of autoimmune encephalitis who presented with acute encephalopathy and seizures. Electroencephalography demonstrated focal seizures superimposed on ictal\u2013interictal continuum activity, and brain MRI revealed cytotoxic lesions of the corpus callosum. High\u2010titer glutamic acid Decarboxylase 65 (GAD65) antibodies supported a relapse of autoimmune encephalitis after other potential etiologies were excluded. The patient demonstrated marked clinical improvement following aggressive immunotherapy, including plasma exchange, high\u2010dose corticosteroids, and rituximab. This case highlights the diagnostic and management challenges of autoimmune encephalitis in patients with APS\u20101, and to our knowledge, represents the first reported case of recurrent autoimmune encephalitis in this population with a favorable response to early immunomodulatory therapy.<\/jats:p>","DOI":"10.1155\/crcc\/6086023","type":"journal-article","created":{"date-parts":[[2026,5,18]],"date-time":"2026-05-18T08:15:21Z","timestamp":1779092121000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Recurrent Autoimmune Encephalitis in a Patient With Autoimmune Polyendocrine Syndrome Type 1"],"prefix":"10.1155","volume":"2026","author":[{"ORCID":"https:\/\/orcid.org\/0009-0004-5380-4843","authenticated-orcid":false,"given":"Jason T.","family":"Stemple","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kamal","family":"Abulebda","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Grace F.","family":"Ryan","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Nadine G.","family":"Haddad","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0009-0002-5419-1298","authenticated-orcid":false,"given":"Alden F.","family":"Dewey","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0009-0008-6381-5430","authenticated-orcid":false,"given":"Jennah C.","family":"Foltz","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4623-1317","authenticated-orcid":false,"given":"Laurence E.","family":"Walsh","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9416-7113","authenticated-orcid":false,"given":"Riad","family":"Lutfi","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2026,5,18]]},"reference":[{"key":"e_1_2_8_1_2","article-title":"Autoimmune Polyendocrinopathy-Candidiasis-Ectodermal Dystrophy (APECED)","volume":"7","author":"Ferr\u00e9 E. 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