![]() The patient was a 37-year-old previously healthy man with a history of primary syphilis who received regular medication with Emtricitabine/Tenofovir as HIV pre-exposure prophylaxis. A recent meta-analysis on neurological and psychiatric presentations of mpox reported seizures, confusion, and encephalitis in < 3% of infected individuals, but the studies have been small and only one included cerebrospinal fluid (CSF) results and neuroimaging. However, the spectrum of severe disease manifestations represents a current knowledge gap. The course of mpox is usually relatively benign and self-limiting, although within the current outbreak around 6% of patients have required hospitalization. Apart from this epidemiological pattern of transmission, cases in the global outbreak frequently show a different clinical presentation from that previously described, with a predominance of anogenital lesions, as well as manifestations such as penile oedema, proctitis, and severe pain from skin and mucosal lesions. Since May 2022, nearly 90,000 cases of mpox have been reported within a global outbreak, with the major proportion of cases affecting men who have sex with men. ![]() This case of mpox-associated encephalitis shows that CNS involvement in mpox infection may have a relatively mild clinical course, and that detection of intrathecal antibody production can be used to establish the diagnosis if CSF monkeypox virus-PCR is negative. He spontaneously improved over a few days course and recovered fully. The CSF was PCR-negative for monkeypox virus but intrathecal antibody production was detected. Neuroimaging and CSF analysis indicated encephalitis. Twelve days later he was admitted with fever and confusion. He first presented with painful skin lesions and genital ulcers polymerase chain reaction (PCR) from the lesions was positive for mpox. We report a previously healthy 37-year-old man with mpox complicated by encephalitis. ![]() Central nervous system (CNS) involvement has previously been described as a rare and severe complication of mpox however, diagnostic findings in cerebrospinal fluid (CSF) analysis and neuroimaging studies have only been reported in one case previously. In the 2022 mpox-outbreak most patients presented with mild symptoms. ![]()
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