A 62-year-old female farmer presented with retinal detachment in her left eye, and an Angiostrongylus cantonensis worm was recovered by vitreous surgery. The case did not show typical clinical symptoms indicating meningitis, although the patient complained of a mild headache, a low-grade fever, and slight ataxia. The symptoms were treated as influenza before the onset of the retinal detachment. The present case is the first confirmed of ocular angiostrongyliasis in Japan.