Human herpesvirus-6 (HHV-6) encephalitis represents a serious opportunistic infection in immunocompromised patients that is associated with substantial mortality and long-term neurological sequelae. 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.

