title: A case of bilateral trochleitis in adult-onset Still's disease.
abstract: OBJECTIVE
  We describe the first case of adult-onset Still's disease (AOSD) presenting with trochleitis and successfully treated with an interleukin-1 receptor antagonist.
  
  
  METHODS
  This is a descriptive case report of one patient, identified in consultation with the inpatient ophthalmology consult service.
  
  
  RESULTS
  A 23-year-old Caucasian male initially presented with complaints of sore throat, myalgias, rash, and fever. After he failed to respond to antibiotic therapy for presumed streptococcal pharyngitis and developed left eye pain with upgaze, he was admitted to the hospital. Laboratory workup revealed elevated inflammatory markers and liver enzymes. He was eventually diagnosed with AOSD. During his hospitalization he developed binocular vertical diplopia, worsening of left eye pain on upgaze, and exquisite tenderness over the region of the left trochlea, consistent with trochleitis. This diagnosis was supported by magnetic resonance imaging (MRI). Soon after onset of left eye symptoms, the right eye developed identical symptoms. He was started on intravenous corticosteroids, which led to the rapid resolution of his ocular and systemic symptoms. However, after transition to oral corticosteroids, his symptoms recurred. He was started on anakinra, an interleukin-1 (IL-1) receptor antagonist, which led to resolution of his ocular and systemic symptoms.
  
  
  CONCLUSIONS
  Trochleitis is an unusual form of orbital inflammation localized to the superior oblique tendon and trochlea complex. It has been associated with a number of systemic inflammatory conditions including systemic lupus erythematous, rheumatoid arthritis, and juvenile idiopathic arthritis. This is the first case of a patient with AOSD presenting with trochleitis. Treatment with IV corticosteroids and anakinra resulted in rapid resolution of his ocular and systemic symptoms.
authors.affiliation: Department of Ophthalmology, The Wilmer Eye Institute, The Johns Hopkins Hospital, 600 N Wolfe St,Wimer B-20, Baltimore, MD 21287. Electronic address: rgutmar1@jhmi.edu.
authors.forename: Ron
authors.initials: R
authors.lastname: Gutmark
authors.affiliation: Department of Ophthalmology, The Wilmer Eye Institute, The Johns Hopkins Hospital, 600 N Wolfe St,Wimer B-20, Baltimore, MD 21287.
authors.forename: Kasra
authors.initials: K
authors.lastname: Eliasieh
authors.affiliation: Department of Ophthalmology, The Wilmer Eye Institute, The Johns Hopkins Hospital, 600 N Wolfe St,Wimer B-20, Baltimore, MD 21287.
authors.forename: Roxana
authors.initials: R
authors.lastname: Rivera-Michlig
mesh_terms: D018501:Antirheumatic Agents / Q000627:therapeutic use*; D005128:Eye Diseases / Q000150:complications* / Q000188:drug therapy; D006801:Humans; D007249:Inflammation / Q000188:drug therapy*; D053590:Interleukin 1 Receptor Antagonist Protein / Q000627:therapeutic use*; D008297:Male; D016706:Still's Disease, Adult-Onset / Q000150:complications* / Q000188:drug therapy; D016896:Treatment Outcome; D055815:Young Adult
chemical_list: D018501:Antirheumatic Agents; D053590:Interleukin 1 Receptor Antagonist Protein
keywords: Adult-onset Still's disease; Anakinra; Interleukin-1 receptor antagonist; Ophthalmology; Superior oblique; Trochleitis