Cavernous malformations (CMs) involving the midbrain are more challenging for surgical treatment than are CMs at other sites because of the surrounding critical structures and deep location. However, specific features and treatment strategies have not been well illustrated.To evaluate the long-term durability of surgical treatment of midbrain CMs (MBCMs) as well as surgical outcomes and complications.A retrospective study was conducted in 34 patients who underwent microsurgical resection of MBCMs between 1995 and 2015. Demographics, lesion characteristics, surgical approaches, surgical outcomes, and complications were analyzed.A total of 34 adult patients with a mean age of 38.6 years were assessed. All patients presented with a history of hemorrhage. Lesion locations included the midbrain (n = 27), midbrain and thalamus (n = 2), and pontomesencephalic junction (n = 5). Mean lesion size was 1.7 cm; average clinical follow-up was 5.6 years. Mean modified Rankin Scale (mRS) scores on admission, at discharge, and at last follow-up were 2.0, 2.7, and 1.7, respectively. Postoperatively, 19 patients (55.9%) showed new or worsened neurologic deficits. Multivariate analysis showed that admission mRS score (≥3) was an independent predictor of poor functional outcome (odds ratio, 50.832; 95% confidence interval, 2.967–901.283; P = 0.007). No rehemorrhage or recurrence case was found during the follow-up period.Although surgery for MBCMs is associated with significant perioperative morbidity and mortality, most patients show favorable outcomes. Higher preoperative mRS score is an independent predictor of poor functional outcome.