A Case of Recurrent Painful Ophthalmoplegic Neuropathy Triggered by Strenuous Activity

Recurrent painful ophthalmoplegic neuropathy (RPON), formerly known as ophthalmoplegic migraine, is a rare condition characterized by recurrent bouts of headache followed by ocular motor nerve palsies. The pathophysiology of RPON remains poorly understood. We report a new case of RPON in which the migraine-like headache is triggered by strenuous activity and exercise, shedding more light on the possible pathogenesis of RPON. A 44-year-old Hispanic male presented to our clinic for his fifth episode of diplopia. He had recurrent episodes of unilateral abducens or oculomotor nerve palsies affecting either eye. He recalled that each episode was immediately preceded by weight-lifting activities, which triggered an ipsilateral headache, followed by ophthalmoplegia 2 days later. Repeated investigations, including blood work, cerebrospinal fluid analysis, Magnetic Resonance Imaging, and Magnetic Resonance Angiography, all returned negative. The ophthalmoplegia eventually resolved with or without corticosteroid treatment. The clinical characteristics of our case suggest that headaches with migraine-like features may remain an important presentation preceding the ophthalmoplegia events in RPON.