Objective: Cavernous malformation(CM) originating from the hypothalamus is extremely rare and the deep location presents a challenge for its neurosurgical management. We report such a case to better understand its clinical features. Methods and Results: A 40-year-old male patient presented with impaired vision in the left eye. Magnetic resonance imaging(MRI) revealed a regularly shaped round lesion located in the suprasellar cistern, and a clinical diagnosis of hypothalamic CM was made. Complete microsurgical excision was performed via a right pterional craniotomy. The patient showed good recovery with no further visual acuity or field deficits postoperatively. No CM recurrence or rebleeding was seen on follow-up MRI scans performed over the course of two years. Conclusions: For patients with cavernous malformation in the hypothalamus, accurate preoperative diagnosis with complete surgical removal by an appropriate surgical approach can contribute to satisfactory outcomes.