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Although incidental human infections have been recognized, comprehensive descriptions remain scarce. Herein, we report the clinical-virological analysis of the first documented autochthonous case of fatal USUV infection in a severely immunocompromised adult from Hungary.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Clinical presentation<\/jats:title>\n                    <jats:p>A 61-year-old female with relapsed acute myelomonocytic leukemia developed progressive neurological symptoms, accompanied by high-grade fever, during post-chemotherapy aplasia. Initial cranial MRI revealed symmetric thalamic and brainstem abnormalities, while cerebrospinal fluid analysis showed mildly elevated protein levels. Despite empirical antimicrobial therapy, her status deteriorated with new-onset dysarthria and somnolence by day\u2009+\u200929 post-chemotherapy, requiring admission to the intensive care unit. Subsequent EEG demonstrated diffuse background slowing, and follow-up MRI confirmed further progression of the lesions. Despite supportive care and extensive microbiological testing, the patient died on day\u2009+\u200937 post-chemotherapy.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Virological investigation<\/jats:title>\n                    <jats:p>\n                      USUV RNA was detected in CSF, blood, urine, and\n                      <jats:italic>post-mortem<\/jats:italic>\n                      tissues by RT-qPCR, using validated\n                      <jats:italic>in-house<\/jats:italic>\n                      protocols. Virus isolation was successfully achieved\n                      <jats:italic>via<\/jats:italic>\n                      intracranial inoculation of newborn mice and subsequent culture in Vero E6 cell cultures. Whole-genome sequencing and phylogenetic analysis confirmed infection with the USUV Europe 2 lineage, closely related to other Hungarian and Italian strains. No other pathogens from the central nervous system were identified.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>We highlight the challenges of USUV infection in immunocompromised patients. The phylogenetic link between European strains shows the regional emergence of high-risk viral lineages. Surveillance, donor screening, and research into antiviral therapies are needed to mitigate the impact of this emerging arbovirus.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1186\/s12985-025-02890-9","type":"journal-article","created":{"date-parts":[[2025,7,29]],"date-time":"2025-07-29T16:45:42Z","timestamp":1753807542000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":13,"title":["First documented case of a fatal autochthonous Usutu virus infection in an immunocompromised patient in Hungary: a clinical-virological report and implications from the literature"],"prefix":"10.1186","volume":"22","author":[{"given":"B\u00e1lint Gergely","family":"Szab\u00f3","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anna","family":"Nagy","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Orsolya","family":"Nagy","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anita","family":"Koroknai","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nikolett","family":"Csonka","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dorina","family":"Kor\u00f3zs","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Krisztina","family":"Jeszenszky","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Apor","family":"Hardi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"N\u00f3ra","family":"De\u00e9zsi-Magyar","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"J\u00e1nos","family":"Sztikler","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zolt\u00e1n","family":"B\u00f3di","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"D\u00e1niel","family":"Cadar","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"G\u00e1bor Endre","family":"T\u00f3th","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lili\u00e1na","family":"Veres","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Erzs\u00e9bet","family":"Barcsay","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"M\u00e1ria","family":"Tak\u00e1cs","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"J\u00e1nos","family":"Sink\u00f3","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2025,7,29]]},"reference":[{"issue":"47","key":"2890_CR1","doi-asserted-by":"publisher","first-page":"1900180","DOI":"10.2807\/1560-7917.ES.2019.24.47.1900180","volume":"24","author":"M Pacenti","year":"2019","unstructured":"Pacenti M, Sinigaglia A, Martello T, de Rui ME, Franchin E, Pagni S, et al. 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