The authors present a first case of cysticercosis in the optic canal in a fifteen-year-old female patient. Cysticercosis of the optic nerve is rare. A cyst in the optic canal, beneath the sheath of the optic nerve, has never been reported. The patient presented with rapidly diminishing vision in the left eye, headache and papillitis. A magnetic resonance imaging revealed a cystic lesion at the entrance of the optic canal. Surgery performed was a transcranial orbitotomy which included deroofing of the optic canal and removal of the cyst from under the sheath of the optic nerve. The cyst proved to be cysticercus histopathologically. The outcome was a remarkable visual recovery.
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