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Midbrain lesions leading to bilateral fourth nerve palsies are typically accompanied by other brainstem symptomatology. Here we report a case of a 29-year-old man with hydrocephalus and significant third ventricle dilation applying pressure on the dorsal midbrain and having as the only manifestation isolated, bilateral fourth cranial nerve palsies. This finding, reported here for the first time, could be attributed to a partially working ventriculoperitoneal shunt previously placed in this patient, which was able to sporadically relieve the increases of the intraventricular pressure on the midbrain that would normally lead to other manifestations.