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The disease most often involves the limbic system, with typical neuroimaging findings restricted to the hippocampal and amygdaloid structures. Here, we describe a rare case of HHV-6 encephalitis presenting with atypical imaging features, which demonstrated near-complete resolution following ganciclovir therapy. This case underscores that HHV-6 encephalitis is not invariably confined to the limbic system and highlights the importance of prompt antiviral therapy in preventing irreversible neurological damage.<\/jats:p>","DOI":"10.47936\/encephalitis.2025.00115","type":"journal-article","created":{"date-parts":[[2026,1,9]],"date-time":"2026-01-09T00:40:12Z","timestamp":1767919212000},"page":"19-23","update-policy":"https:\/\/doi.org\/10.47936\/crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Atypical manifestation of human herpes virus-6 encephalitis with extra-limbic lesions: a case report"],"prefix":"10.47936","volume":"6","author":[{"ORCID":"https:\/\/orcid.org\/0009-0004-0434-9223","authenticated-orcid":true,"given":"Soo Hyun","family":"Ahn","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Seon-Jae","family":"Ahn","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5863-0302","authenticated-orcid":true,"given":"Kon","family":"Chu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"27912","reference":[{"key":"ref1","doi-asserted-by":"publisher","DOI":"10.1016\/j.medmal.2016.09.004"},{"key":"ref2","doi-asserted-by":"publisher","DOI":"10.1038\/bmt.2017.175"},{"key":"ref3","doi-asserted-by":"publisher","DOI":"10.1093\/ofid\/ofz269"},{"key":"ref4","doi-asserted-by":"publisher","DOI":"10.1212\/nxi.0000000000000942"},{"key":"ref5","doi-asserted-by":"publisher","DOI":"10.7759\/cureus.23007"},{"key":"ref6","doi-asserted-by":"publisher","DOI":"10.4103\/aian.aian_717_24"},{"key":"ref7","doi-asserted-by":"publisher","DOI":"10.1093\/ofid\/ofae501"},{"key":"ref8","doi-asserted-by":"publisher","DOI":"10.3324\/haematol.2019.223073"},{"key":"ref9","first-page":"887","volume-title":"Mr imaging of human herpesvirus-6-associated encephalitis in 4 patients with anterograde amnesia after allogeneic hematopoietic stem-cell transplantation","author":"Gorniak","year":"2006"},{"key":"ref10","doi-asserted-by":"publisher","DOI":"10.2214\/AJR.09.2548"},{"key":"ref11","doi-asserted-by":"publisher","DOI":"10.1016\/j.pediatrneurol.2009.07.014"},{"key":"ref12","doi-asserted-by":"publisher","DOI":"10.1148\/radiol.2492071917"},{"key":"ref13","doi-asserted-by":"publisher","DOI":"10.1016\/j.jtct.2025.04.016"},{"key":"ref14","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365-2141.2003.04788.x"},{"key":"ref15","doi-asserted-by":"publisher","DOI":"10.1038\/s41409-024-02398-w"},{"key":"ref16","doi-asserted-by":"publisher","DOI":"10.1016\/s1386-6532(06)70012-9"},{"key":"ref17","doi-asserted-by":"publisher","DOI":"10.1111\/tid.13443"}],"container-title":["encephalitis"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/encephalitisjournal.org\/upload\/pdf\/encephalitis-2025-00115.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/encephalitisjournal.org\/journal\/view.php?doi=10.47936\/encephalitis.2025.00115","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/encephalitisjournal.org\/upload\/pdf\/encephalitis-2025-00115.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,1,9]],"date-time":"2026-01-09T00:40:16Z","timestamp":1767919216000},"score":1,"resource":{"primary":{"URL":"http:\/\/encephalitisjournal.org\/journal\/view.php?doi=10.47936\/encephalitis.2025.00115"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,1,10]]},"references-count":17,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2026,1]]}},"URL":"https:\/\/doi.org\/10.47936\/encephalitis.2025.00115","relation":{},"ISSN":["2765-4559","2734-1461"],"issn-type":[{"value":"2765-4559","type":"print"},{"value":"2734-1461","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,1,10]]},"assertion":[{"value":"2025-10-11","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-11-17","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-01-10","order":2,"name":"published","label":"Published online","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-01-10","order":3,"name":"published","label":"Print publication","group":{"name":"publication_history","label":"Publication History"}}]}}