title: Bilateral Ocular Myositis Associated with Whipple's Disease.
abstract: OBJECTIVE
  To describe the clinical features of a Caucasian female patient with a history of treated gastrointestinal Whipple's disease (WD) who developed new-onset diplopia, with a description of the histopathological features of the extraocular muscle biopsies.
  
  
  METHODS
  A previously fit 38-year-old Caucasian female presented with acute-onset diplopia after being on a sustained medication regime for biopsy-proven gastrointestinal WD. A magnetic resonance imaging scan of her orbits with gadolinium revealed diffuse enhancement of the bellies of the extraocular muscles bilaterally, particularly the medial rectus, superior rectus, and superior oblique muscles, consistent with an infiltrative myositis. She underwent unilateral extraocular muscle biopsies.
  
  
  RESULTS
  The extraocular muscle biopsies contained macrophages between the muscle fibres. These contained periodic acid-Schiff-positive cytoplasmic granules. Immunohistochemistry with an antibody raised toTropheryma whippleishowed positive staining of the same macrophages. Transmission electron microscopy confirmed the presence of effeteT. whippleicell membranes in lysosomes.
  
  
  CONCLUSIONS
  This case describes bilateral WD-associated extraocular muscle myositis. The exact mechanism for this unusual presentation is unclear, but both a WD-associated immune reconstitution inflammatory syndrome and treatment failure are possibilities, with a good response observed to antibiotic therapy and adjunctive corticosteroids.
authors.affiliation: Department of Infection and Tropical Medicine, Royal Hallamshire Hospital, Sheffield, UK.
authors.forename: Vivak
authors.initials: V
authors.lastname: Parkash
authors.affiliation: National Specialist Ophthalmic Pathology Service (NSOPS), Department of Histopathology, Royal Hallamshire Hospital, Sheffield, UK.
authors.forename: Hardeep Singh
authors.initials: HS
authors.lastname: Mudhar
authors.affiliation: Electron Microscopy Unit, Department of Histopathology, Royal Hallamshire Hospital, Sheffield, UK.
authors.forename: Bart E
authors.initials: BE
authors.lastname: Wagner
authors.affiliation: Aix-Marseille Université, Unité de Recherche sur les Maladies Infectieuses et Tropicales Emergentes, Marseille, France.
authors.forename: Didier
authors.initials: D
authors.lastname: Raoult
authors.affiliation: Department of Radiology, Royal Hallamshire Hospital, Sheffield, UK.
authors.forename: Ruth
authors.initials: R
authors.lastname: Batty
authors.affiliation: Aix-Marseille Université, Unité de Recherche sur les Maladies Infectieuses et Tropicales Emergentes, Marseille, France.
authors.forename: Hubert
authors.initials: H
authors.lastname: Lepidi
authors.affiliation: Department of Ophthalmology, Royal Hallamshire Hospital, Sheffield, UK.
authors.forename: John
authors.initials: J
authors.lastname: Burke
authors.affiliation: Department of Infection and Tropical Medicine, Royal Hallamshire Hospital, Sheffield, UK; Department of Infection, Immunity and Cardiovascular Disease, The Medical School, University of Sheffield, Sheffield, UK.
authors.forename: Paul
authors.initials: P
authors.lastname: Collini
authors.affiliation: Department of Infection and Tropical Medicine, Royal Hallamshire Hospital, Sheffield, UK; Department of Infection, Immunity and Cardiovascular Disease, The Medical School, University of Sheffield, Sheffield, UK.
authors.forename: Thushan
authors.initials: T
authors.lastname: de Silva
keywords: Extraocular muscle; Immune reconstitution inflammatory syndrome; Ocular myositis; Whipple's disease