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Recurrent painful ophthalmoplegic neuropathy (RPON) is a rare neurological disorder characterized by recurring ipsilateral headache and paresis of one or more ocular motor nerves. We report the case of a 56-year-old woman with systemic lupus erythematosus (SLE) and hypertension, who presented with severe recurring headaches, nausea, and vomiting. Initially misdiagnosed with cerebral venous sinus thrombosis, her symptoms persisted despite anticoagulant therapy. Further evaluation led to the diagnosis of RPON. Management included intravenous analgesia, hydration, and indomethacin for pain relief. Persistent headache episodes necessitated the introduction of lamotrigine, resulting in significant symptom improvement. However, discontinuation of lamotrigine led to a recurrence of symptoms, which resolved upon resuming the medication. This case contributes to the limited RPON literature, providing insights into its diagnosis and management, with the goal of enhancing awareness and improving patient care., Competing Interests: The authors have declared that no competing interests exist., (Copyright © 2024, Aljammaz et al.)

Recurrent painful ophthalmoplegic neuropathy (RPON), previously known as ophthalmoplegic migraine, is a disorder typically characterized by recurrent episodes of unilateral headache concurrent with ipsilateral ocular cranial nerve paresis which primarily affects children. Diagnosis is mostly one of exclusion, based on clinical symptoms, supplemented by imaging for enhanced or distorted oculomotor nerves. We present a case of RPON in a 24-year-old adult female with unique features of unilateral left headache with ipsilateral pupillary dilation spreading to bilateral dilation and no MRI findings of oculomotor nerve enhancement., Competing Interests: The authors have declared that no competing interests exist., (Copyright © 2024, Koo et al.)