| Summary: | Here, we describe an extraordinary case of a 38-year-old male patient with five recurrent episodes of ophthalmoplegia preceded by unilateral migraine-like headaches. He reported recurrent episodes of third cranial nerve palsy preceded by a pulsating and throbbing headache with mild photophobia for 48 hours responsive to steroid therapy, and this was his fifth attack. The clinical symptoms resolved completely after steroid therapy. After a detailed differential diagnosis was made to exclude alternative diagnoses, the patient was finally diagnosed as having recurrent painful ophthalmoplegic neuropathy (RPON). Two-thirds of RPON cases are monophasic. The underlying pathophysiologic mechanisms of RPON are still unclear, however, different hypotheses such as ischemia, demyelination, inflammation, or compression of the nerve are suggested. It is important to be alert for RPON in cases of recurrent ophthalmoplegia and make a detailed differential diagnosis.
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