Tolosa–Hunt syndrome presented with painless and fluctuating ophthalmoplegia

The diagnosis of Tolosa–Hunt syndrome is typically based on the presence of painful ophthalmoplegia, magnetic resonance imaging (MRI) findings, exclusion of other causes, and a rapid response to steroid treatment. We report a rare case of Tolosa–Hunt syndrome with atypical presenting features, including painless and variable ophthalmoplegia which resolved spontaneously. An MRI scan at that time was considered normal. However, 2 months later, the patient had a second episode characterized with painful ophthalmoplegia and progressive blurry vision OD. Repeated MRI studies revealed an enhanced lesion in the right orbital apex and cavernous sinus. The patient responded well to corticosteroid treatment. Patients with Tolosa–Hunt syndrome could initially present with painless and fluctuating ophthalmoplegia. A careful review of the MRI scans is recommended. Painless Tolosa–Hunt syndrome should be considered in the differential diagnosis when initial investigations for other causes, including myasthenia gravis, are negative.