Parkash, V., Mudhar, H.S., Wagner, B.E. et al. (6 more authors) (2016) Bilateral Ocular Myositis Associated with Whipple's Disease. Ocular Oncology and Pathology, 3. pp. 17-21. ISSN 2296-4681
Abstract
Purpose: 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 to Tropheryma whipplei showed positive staining of the same macrophages. Transmission electron microscopy confirmed the presence of effete T. whipplei cell membranes in lysosomes.
Conclusion: 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.
Metadata
Item Type: | Article |
---|---|
Authors/Creators: |
|
Copyright, Publisher and Additional Information: | © Karger Publishers. This is the peer-reviewed but unedited manuscript version of the following article: Parkash V, Mudhar H, S, Wagner B, E, Raoult D, Batty R, Lepidi H, Burke J, Collini P, de Silva T, Bilateral Ocular Myositis Associated with Whipple's Disease. Ocul Oncol Pathol 2017;3:17-21. The final, published version is available at https://doi.org/10.1159/000448622 |
Keywords: | Whipple’s disease ; Ocular myositis ; Extraocular muscle ; Immune reconstitution inflammatory syndrome |
Dates: |
|
Institution: | The University of Sheffield |
Academic Units: | The University of Sheffield > Faculty of Medicine, Dentistry and Health (Sheffield) > Department of Infection, Immunity and Cardiovascular Disease The University of Sheffield > Sheffield Teaching Hospitals |
Depositing User: | Symplectic Sheffield |
Date Deposited: | 29 Jun 2017 14:07 |
Last Modified: | 04 Nov 2017 23:54 |
Published Version: | https://doi.org/10.1159/000448622 |
Status: | Published |
Publisher: | Karger Publishers |
Refereed: | Yes |
Identification Number: | 10.1159/000448622 |
Open Archives Initiative ID (OAI ID): | oai:eprints.whiterose.ac.uk:118218 |