Tolosa-Hunt Syndrome with Multiple Orbital Myositis in a Patient with Crohn's Disease

Purpose: We report a case of Tolosa-Hunt syndrome with multiple orbital myositis identified via orbital magnetic resonance imaging in a patient with Crohn's disease who developed right eye pain and binocular horizontal diplopia. Case summary: A 46-year-old woman visited our clinic with a 2-month history of right eye pain and migraine, as well as a 3-day history of acute horizontal diplopia. She had previously been diagnosed with Crohn's disease and was taking immunosuppressive drugs. In the eye movement test, esotropia and an abduction limitation of -3.0 in the right eye were observed on the Krimsky test. There were no specific findings in anterior segment and fundus examinations. Orbital magnetic resonance imaging showed multiple extraocular muscle enhancement in the right eye and multiple extraocular muscle hypertrophy in the left eye. The patient was diagnosed with binocular multiple orbital myositis and right Tolosa-Hunt syndrome; she was treated with high-dose steroids for 3 days followed by lower dose oral medications. During the first week of treatment, the right eye pain disappeared and the right eye abduction limitation showed slight improvement. After 3 months of treatment, the right eye abduction limitation and esotropia completely disappeared. Conclusions: Orbital myositis and Tolosa-Hunt syndrome are idiopathic, nonspecific chronic granulomatous diseases with painful ophthalmoplegia. We describe a rare case in which the two diseases appear together.