BACKGROUND: Human immunodeficiency virus (HIV) infection is linked with an uncommon vasculopathy syndrome, increasing the susceptibility of infected individuals to develop aneurysms across systemic vasculature, notably in the cerebral vasculature. Intracranial aneurysms have been detected in up to 14% of HIV-positive patients with neuroimaging, often manifesting in unusual locations or with atypical morphologies due to systemic pathophysiology. OBSERVATIONS: This case report describes a previously coiled middle cerebral artery sidewall aneurysm that subsequently recurred in an HIV-positive man in his late 20s, necessitating open treatment with microsurgical clip reconstruction, which was performed using a minipterional craniotomy and transsylvian approach. Intraoperative findings included diffuse cerebral dolichoectasia, a broad-necked recurrence of the M2 segment aneurysm, and a de novo A1 segment aneurysm. Both aneurysms were successfully treated with primary clipping reconstruction. The patient recovered well, with no new postoperative neurological deficits and radiographically confirmed obliteration of both aneurysms. LESSONS: Endovascular treatment of HIV-positive patients with cerebrovascular disease can be predisposed to failure, and such patients require close radiographic surveillance. The differential risk of recurrence by treatment modality is indeterminate
however, young HIV-positive patients with intracranial aneurysms can benefit from the preferential use of open clipping where equipoise exists. https://thejns.org/doi/10.3171/CASE24700.