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A man aged 57 years presented to the emergency department after 3 weeks of bilateral periorbital pain and swelling with associated headaches. He reported diplopia and pain with extraocular movement. There were no constitutional, dermatologic, or neurologic symptoms. The patient endorsed a history of Crohn disease, and his medication had been changed from infliximab to ustekinumab 6 weeks prior owing to persistent intestinal inflammation.
Clinical examination revealed bilateral periorbital edema, diplopia with decreased abduction in the right eye and decreased adduction in the left eye, chemosis, proptosis, and conjunctival hyperemia (Figure 1A). Visual acuity and neurologic and fundus examination findings were otherwise normal with no sign of optic neuropathy.
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Medication-induced orbital myositis secondary to ustekinumab
B. Change therapies for Crohn disease
The patient had features of a corticosteroid-responsive bilateral orbital myositis (OM). OM was first described in 1903.1 It can occur at any age and more commonly affects women.2 It commonly presents as painful diplopia or orbital inflammation with chemosis, proptosis, or conjunctival hyperemia. The horizontal recti muscles are most frequently affected, although the superior or inferior recti can also be involved.3 The inflammatory process (Figure 2) appears to be mediated by helper T-cell type 1, with significant elevations in interleukin 12, tumor necrosis factor α, and interferon γ.4 Diagnosis of OM is based on physical examination and radiologic features excluding other etiologies and may require tissue biopsy when features are atypical or response to corticosteroids is inadequate.
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CME Disclosure Statement: Unless noted, all individuals in control of content reported no relevant financial relationships. If applicable, all relevant financial relationships have been mitigated.
Corresponding Author: Abhinav Vasudevan, BMedicine, MPH, PhD, Division of Gastroenterology and Hepatology, Mayo Clinic, 200 1st St SW, Rochester, MN 55905 (email@example.com).
Published Online: January 27, 2022. doi:10.1001/jamaophthalmol.2021.4676
Conflict of Interest Disclosures: Dr Vasudevan reported receiving nonfinancial support from Ferring Pharmaceuticals to attend an educational conference. No other disclosures were reported.
Additional Contributions: We thank the patient for granting permission to publish this information.
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