Article ; Online: Sudden unexpected death in epilepsy and ictal asystole in patients with autoimmune encephalitis: a systematic review.
2024 Volume 45, Issue 6, Page(s) 2811–2823
Abstract: Objective: As autoimmune encephalitis (AE) often involves the mesial temporal structures which are known to be involved in both sudden unexpected death in epilepsy (SUDEP) and ictal asystole (IA), it may represent a good model to study the ... ...
| Abstract | Objective: As autoimmune encephalitis (AE) often involves the mesial temporal structures which are known to be involved in both sudden unexpected death in epilepsy (SUDEP) and ictal asystole (IA), it may represent a good model to study the physiopathology of these phenomena. Herein, we systematically reviewed the occurrence of SUDEP and IA in AE. Methods: We searched 4 databases (MEDLINE, Scopus, Embase, and Web of Science) for studies published between database inception and December 20, 2022, according to the PRISMA guidelines. We selected articles reporting cases of definite/probable/possible/near-SUDEP or IA in patients with possible/definite AE, or with histopathological signs of AE. Results: Of 230 records assessed, we included 11 cases: 7 SUDEP/near-SUDEP and 4 IA. All patients with IA were female. The median age at AE onset was 30 years (range: 15-65), and the median delay between AE onset and SUDEP was 11 months; 0.9 months for IA. All the patients presented new-onset seizures, and 10/11 also manifested psychiatric, cognitive, or amnesic disorders. In patients with SUDEP, 2/7 were antibody-positive (1 anti-LGI1, 1 anti-GABABR); all IA cases were antibody-positive (3 anti-NMDAR, 1 anti-GAD65). Six patients received steroid bolus, 3 intravenous immunoglobulin, and 3 plasmapheresis. A pacemaker was implanted in 3 patients with IA. The 6 survivors improved after treatment. Discussion: SUDEP and IA can be linked to AE, suggesting a role of the limbic system in their pathogenesis. IA tends to manifest in female patients with temporal lobe seizures early in AE, highlighting the importance of early diagnosis and treatment. |
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| MeSH term(s) | Humans ; Sudden Unexpected Death in Epilepsy ; Encephalitis/complications ; Encephalitis/physiopathology ; Heart Arrest/complications ; Heart Arrest/mortality ; Hashimoto Disease/complications ; Hashimoto Disease/physiopathology ; Female ; Adolescent ; Adult ; Epilepsy/complications ; Epilepsy/mortality ; Epilepsy/physiopathology ; Young Adult ; Middle Aged |
| Language | English |
| Publishing date | 2024-01-09 |
| Publishing country | Italy |
| Document type | Systematic Review ; Journal Article |
| ZDB-ID | 2016546-8 |
| ISSN | 1590-3478 ; 1590-1874 |
| ISSN (online) | 1590-3478 |
| ISSN | 1590-1874 |
| DOI | 10.1007/s10072-023-07280-z |
| Database | MEDical Literature Analysis and Retrieval System OnLINE |
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