Neurocysticercosis is a common tropical infection presenting with neurological signs. It commonly presents as seizures but various other focal neurological presentations have been reported. Though neurocysticercosis have been reported to present as isolated internuclear ophthalmoplegia, we report the first case of neurocysticercosis presenting as wall-eyed monoocular internuclear ophthalmoplegia syndrome with contraversive ocular tilt reaction.