OBJECTIVE The endoscopic fenestration of intraventricular CSF cysts has evolved into a well-accepted treatment modality. However, definition of the optimal trajectory for endoscopic fenestration may be difficult. Distorted ventricular anatomy and poor visibility within the cyst due to its contents can make endoscopic fenestration challenging if approached from the ipsilateral side. In addition, transcortical approaches can theoretically cause injury to eloquent cortex, particularly in patients with dominant-sided lesions. The aim of this study was to examine the value of the contralateral transcortical transventricular approach in patients with dominant-sided ventricular cysts. METHODS During a 5-year period between 2007 and 2011, 31 patients with intraventricular CSF cysts underwent surgery by the senior author (R.R.). Fourteen of these patients had cysts located on the dominant side. An image-guided endoscopic cyst fenestration via the contralateral transcortical transventricular approach was performed in 11 patients. A retrospective chart review was performed in all these patients to extract data on clinical presentation, operative technique, and surgical outcome. RESULTS The most common presenting symptom was headache, followed by memory deficits and cognitive deterioration. In all cases CSF cysts were space occupying, with associated obstructive hydrocephalus in 8 patients. Image-guided endoscopic fenestration was successfully performed in all cases, with septum pellucidotomy necessary in 6 cases, and endoscopic third ventriculostomy in 1 case for additional aqueductal occlusion. Postoperative clinical outcome was excellent, with no associated permanent neurological or neuropsychological morbidity. No recurrent cysts were observed over a mean follow-up period of 2 years and 3 months. CONCLUSIONS The contralateral approach to ventricular cysts can achieve excellent surgical outcomes while minimizing approach-related trauma to the dominant hemisphere. Careful case selection is essential to ensure that the contralateral endoscopic trajectory is the best possible exposure for sufficient cyst fenestration and restoration of CSF circulation.