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Sahara Hospital, Department of Neurosurgery, Lucknow, India DOI : 10.5137/1019-5149.JTN.2766-09.1 A 43-year-old male presented with 3-month history of low-grade fever and headache. Radiological investigations revealed unilateral hydrocephalus. Unilateral obstruction of the foramen of Monro due to chronic tubercular ependymal inflammation was suspected and endoscopic septostomy was planned. Though ventriculo-peritoneal shunt is a simple method to treat hydrocephalus, complications related to this procedure are numerous. Neuroendoscopy is a safe method to treat hydrocephalus in selected cases, and also provides access to biopsy the lesion in question. An isolated tuberculoma obstructing the foramen of Monro was seen during endoscopy. Presentation and management of this unusual tuberculoma is reported along with a review of the pertinent literature. Keywords : Unilateral hydrocephalus, Tuberculoma, Foramen of monro, Septostomy, Unusual presentation