Orbital manifestation of whipple's disease: An atypical case

Lieger, Olivier; Otto, Sven; Clemetson, I.; Arnold, Marlène; Iizuka, Tetsuya

doi:10.1016/j.jcms.2007.03.005

Cited by 14 publications

(6 citation statements)

References 13 publications

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“…However, in the case of pseudotumor orbitae, more aggressive steps such as surgical decompression may be required to prevent ocular damage and visual loss. 82 In conclusion, Whipple's disease is a systemic illness with diverse clinical presentations, a minority of which may include the skin. Cutaneous Whipple's disease can be classified into those disorders related to malnutrition and those related directly to T whipplei infection, manifested as inflammatory responses to viable T whipplei or its antigens.…”

Section: Discussionmentioning

confidence: 99%

Erythema nodosum–like lesions in treated Whipple's disease: Signs of immune reconstitution inflammatory syndrome

Schaller¹

2009

Journal of the American Academy of Dermatology

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Section: Discussionmentioning

confidence: 99%

Erythema nodosum–like lesions in treated Whipple's disease: Signs of immune reconstitution inflammatory syndrome

Schaller¹

2009

Journal of the American Academy of Dermatology

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“…Other patterns of ocular involvement have been described with WD, similar to many infectious diseases [8]. Numerous reported secondary manifestations of eye disease in WD include uveitis, retinitis, papilloedema, and optic atrophy [9,10,11].…”

Section: Discussionmentioning

confidence: 99%

Bilateral Ocular Myositis Associated with Whipple's Disease

et al. 2016

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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.

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“…Although the literature generally supports antibiotic prophylaxis in all cases of orbital emphysema, this is of unproven benefit and has evolved through convention and personal preference (Silver et al, 1992;Lieger et al, 2007). The enhanced risk of microbial spread with forced airflow across the sino-orbital communication has lead to several reported cases of orbital cellulitus (Silver et al, 1992).…”

Section: Discussionmentioning

confidence: 99%