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We report the first case of subtemporal keyhole surgery approach with neuronavigation and endoscopy, for total resection of an uncommon case of epidermoid cyst of the Meckel’s cave in a 43-year-old woman who presented with paroxysmal headache and diplopia. Magnetic resonance imagines revealed the tumor location on the right area of the cavernous sinus. Postoperative examinations showed complete recovery of symptoms.