A 43-year-old woman suffered from bilateral uveitis and was suspected of sarcoidosis. General fatigue slowly progressed, and urinary incontinence and disturbance of consciousness also developed. When she was admitted to our hospital by ambulance, her conciousness was disturbed, GCS 3-4-6. Enlargement of all ventricles was shown and cell count was moderately elevated. She underwent VP shunt and fully recovered. However, her activity become disturbed again and, after five months, only her left lateral ventricle dilated. We presumed it was caused by the occlusion of the foramen of Monro. Neuroendoscopic inspection of the right lateral ventricle via a left precoronal burrhole revealed a thick, yellish membrane that occluded the foramen of Monro. She received a left VP shunt and cured. She was diagnosed as having recurrent hydrocepalus due to the uveo-meningeal syndrome. We present this first case and discuss this syndrome associated hydrocephalus.