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The intraventricular location of a cavernoma is a rare entity and accounts for approximately 2.5% of all cavernomas of the central nervous system. They are commonly found in the lateral ventricle followed by the third and fourth ventricles. The location in the septum pellucidum is rare, and only four cases have been reported in the international literature. An open craniotomy was performed in all these cases. To the best of our knowledge, this is the first case of a cavernoma of the septum pellucidum successfully resected using a purely endoscopic transventricular approach.

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