Headache and Pain Research

Case Report

Early Infantile Onset of Presumed Recurrent Painful Ophthalmoplegic Neuropathy: Kristen M. Yang, Robert H. Fryer; Received July 6, 2026 Accepted August 12, 2026 Published online October 19, 2026; DOI: https://doi.org/10.62087/hpr.2026.0032 [Epub ahead of print]

Abstract PDF: Recurrent painful ophthalmoplegic neuropathy (RPON) is a rare recurrent cranial neuropathy that usually presents in childhood, most often before 10 years of age. Although RPON primarily affects pediatric patients, onset before 1 year of age is exceedingly rare. We report the case of a 10-month-old boy who presented with recurrent episodes of disconjugate gaze and left-sided ptosis, leading to a presumptive diagnosis of RPON. Magnetic resonance imaging (MRI) showed enhancement of the left oculomotor nerve, a characteristic finding of RPON. To our knowledge, this is one of the youngest reported cases of presumed RPON with oculomotor nerve enhancement on MRI. This case highlights the importance of including RPON in the differential diagnosis of acute ophthalmoplegia in infants, outlines key diagnostic considerations, and compares the patient’s clinical and radiographic findings with those reported in previously published pediatric cases.

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