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Orbital Inflammation in an Adult With Crohn Disease

Educational Objective
Based on this clinical scenario and the accompanying image, understand how to arrive at a correct diagnosis.
1 Credit CME

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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Article Information

Corresponding Author: Abhinav Vasudevan, BMedicine, MPH, PhD, Division of Gastroenterology and Hepatology, Mayo Clinic, 200 1st St SW, Rochester, MN 55905 (abhinav.vasudevan@monash.edu).

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.

References
1.
Gleason  JE .  Idiopathic myositis involving the extraocular muscles.   Ophthalmic Rec. 1903;12:471-478.Google Scholar
2.
Siatkowski  RM , Capó  H , Byrne  SF ,  et al.  Clinical and echographic findings in idiopathic orbital myositis.   Am J Ophthalmol. 1994;118(3):343-350. doi:10.1016/S0002-9394(14)72959-9PubMedGoogle ScholarCrossref
3.
Costa  RM , Dumitrascu  OM , Gordon  LK .  Orbital myositis: diagnosis and management.   Curr Allergy Asthma Rep. 2009;9(4):316-323. doi:10.1007/s11882-009-0045-yPubMedGoogle ScholarCrossref
4.
Wladis  EJ , Iglesias  BV , Gosselin  EJ .  Characterization of the molecular biologic milieu of idiopathic orbital inflammation.   Ophthalmic Plast Reconstr Surg. 2011;27(4):251-254. doi:10.1097/IOP.0b013e31820768f7PubMedGoogle ScholarCrossref
5.
Mannor  GE , Rose  GE , Moseley  IF , Wright  JE .  Outcome of orbital myositis. clinical features associated with recurrence.   Ophthalmology. 1997;104(3):409-413. doi:10.1016/S0161-6420(97)30300-5PubMedGoogle ScholarCrossref
6.
Garrity  JA , Coleman  AW , Matteson  EL , Eggenberger  ER , Waitzman  DM .  Treatment of recalcitrant idiopathic orbital inflammation (chronic orbital myositis) with infliximab.   Am J Ophthalmol. 2004;138(6):925-930. doi:10.1016/j.ajo.2004.06.077PubMedGoogle ScholarCrossref
7.
Lakatos  L , Pandur  T , David  G ,  et al.  Association of extraintestinal manifestations of inflammatory bowel disease in a province of western Hungary with disease phenotype: results of a 25-year follow-up study.   World J Gastroenterol. 2003;9(10):2300-2307. doi:10.3748/wjg.v9.i10.2300PubMedGoogle ScholarCrossref
8.
McNab  AA .  Orbital myositis: a comprehensive review and reclassification.   Ophthalmic Plast Reconstr Surg. 2020;36(2):109-117. doi:10.1097/IOP.0000000000001429PubMedGoogle ScholarCrossref
9.
Pirbhai  A , Rajak  SN , Goold  LA ,  et al.  Bisphosphonate-induced orbital inflammation: a case series and review.   Orbit. 2015;34(6):331-335. doi:10.3109/01676830.2015.1078380PubMedGoogle ScholarCrossref
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US Food and Drug Administration. FDA Adverse Events Reporting System (FAERS) Public Dashboard. Updated October 22, 2021. Accessed September 12, 2021. https://www.fda.gov/drugs/questions-and-answers-fdas-adverse-event-reporting-system-faers/fda-adverse-event-reporting-system-faers-public-dashboard
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