To describe a case of recurrent Tolosa-Hunt syndrome presenting as painful ophthalmoplegia involving different ipsilateral cranial nerves.

Tolosa-Hunt syndrome (THS) is a rare condition characterized by painful ophthalmoplegia due to inflammation of the cavernous sinus or superior orbital fissure, typically involving cranial nerves III, IV, and VI. Recurrent episodes are uncommon, and diagnosis is based on exclusion of other etiologies such as tumors, vascular lesions, and infections. Treatment with corticosteroids often leads to symptom improvement.

A 35-year-old Hispanic male with a prior history of right CN VI palsy presented with headache and diplopia. The headache began two weeks prior, localized to the right occipital region with throbbing pain around the right periorbital area. He was initially treated with a 7-day course of steroids at an outside hospital, which alleviated the headache but not the diplopia. Upon completion of steroids, his headache returned, prompting him to seek further evaluation. Examination revealed right CN IV palsy with painful ophthalmoplegia. Extensive workup, including MRI of the brain and orbits and CT of the thorax, was negative for abnormalities, ruling out mass lesions or sarcoidosis. Lumbar puncture was also normal. He was diagnosed with recurrent Tolosa-Hunt syndrome and discharged on a tapering course of steroids. At follow-up two months later, his symptoms had resolved. One year earlier, he presented with similar complaints of headache and diplopia, with negative workup results, including CT and MRI, leading to a diagnosis of Tolosa-Hunt syndrome and successful treatment with steroids.

This case highlights recurrent Tolosa-Hunt syndrome, presenting with painful ophthalmoplegia and involvement of different cranial nerves. While rare, recurrent episodes of Tolosa-Hunt syndrome should be considered in similar presentations. Corticosteroids remain an effective treatment, providing significant symptom relief.