Recurrent Orbital Inflammation and Whipple Disease

Huerva, Valentín; Espinet, Ramón; Galindo, Carlos

doi:10.1080/09273940801899756

Cited by 7 publications

(5 citation statements)

References 3 publications

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“…Extragastrointestinal manifestations of WD, although uncommon, can encompass cardiovascular, musculoskeletal, and central and peripheral nervous system involvement. Ocular involvement and myositis are rare manifestations of WD, with only 2 previous cases of unilateral ocular myositis described in the literature [4,5]. Generalised myositis has, however, been reported as a result of WD in other cases [6].…”

Section: Discussionmentioning

confidence: 99%

“…An alternative possibility is that myositis was a late presentation of the disease [4,5], occurring as part of an immune reconstitution inflammatory syndrome (IRIS). In a large proportion of WD-associated IRIS cases, however, patients had complete resolution of the symptoms before a late recurrence of inflammation, in contrast to this case [18].…”

Section: Discussionmentioning

confidence: 99%

“…Most WD-associated IRIS cases, however, tend to show a more generalised inflammatory picture, and treatment failures on co-trimoxazole are well recognised. It is noteworthy that both previously described cases of ocular myositis occurred after over 1 year of antibiotic therapy for WD [4,5], with one case requiring corticosteroids in addition to further antibiotic therapy to achieve resolution [4]. The use of steroids, however, in WD-associated IRIS has a limited evidence base, in contrast to steroid use in tuberculosis-associated IRIS [20].…”

Section: Discussionmentioning

confidence: 99%

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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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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

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Bilateral Ocular Myositis Associated with Whipple's Disease

et al. 2016

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“…Imaging characteristics demonstrate enlargement of the lacrimal gland and extraocular muscles. Some of the published reports indicate that the orbital inflammation is secondary to the initial infection with T. Whipplei; however, others indicate that some of the presentations to represent immune reconstitution syndrome (IRIS) in Whipple's disease, particularly in patients who have already had an initial clinical response to effective antimicrobial treatment, have had recurrence of inflammation and who have had negative histologic and molecular evidence of ongoing infection [53][54][55][56]. Because the orbital manifestation of primary Whipple's disease and IRIS can overlap, it can be difficult to determine which cause is driving the inflammatory response.…”

Section: Orbitalmentioning

confidence: 99%

“…Histologically, PAS-positive inclusions are identified in involved macrophages in the tissue. In patients with neurologic involvement, PAS staining of the CNS has been noted to be present in 95% of cases, some of which had negative PAS staining on bowel biopsy [56]. PAS positivity however can be seen with other bacterial infections including M. avium complex and therefore is not specific for Whipple's disease [13].…”

Section: Clinical Diagnosismentioning

confidence: 99%

Ophthalmic manifestations of Whipple's disease

Rasool

2023

Current Opinion in Ophthalmology

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Purpose of review Whipple's disease is an infectious cause of uveitis that may present with nonspecific findings of intraocular inflammation, which can precede the development of neurologic symptoms and signs. Whipple's disease, then, may evade consideration in the differential diagnosis for uveitis. Recent findings Molecular tests can be helpful in identifying the presence of Tropheryma whipplei from ocular specimens. The application of metagenomic sequencing for ocular specimens is promising, as it offers the opportunity to identify the pathogen when suspicion for an intraocular infection is high. Whipple's disease demonstrates the ability to abrogate the host immune response, which gives some insight into its pathogenesis. Summary Whipple's disease should be suspected in patients who have uveitis refractory to anti-inflammatory therapy. Knowledge of this important pathogen can help direct the timely implementation of diagnostic testing.

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Ocular Myositis

Fraser

Skalicky

Gurbaxani

et al. 2012

Curr Allergy Asthma Rep

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Ocular myositis frequently manifests with orbital pain and diplopia. The diagnosis of ocular myositis falls within the overall classification of idiopathic orbital inflammatory diseases, defined as non-infective non-specific orbital inflammation without identifiable local or systemic causes. Orbital myositis may form part of more widespread systemic inflammatory processes such as Crohn's disease and the more recently described IgG4-related disease. There is also a broad range of ophthalmic differential diagnoses. Diagnosis, assessment and management of ocular myositis requires the cooperation of ophthalmologists and rheumatologists/immunologists in order to achieve the best patient outcomes. The current literature and avenues of future research are reviewed.

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Recurrent Orbital Inflammation and Whipple Disease

Abstract: Recurrence of duodenal Whipple disease may be associated with an OI picture that is immmunologically mediated.

Cited by 7 publications

References 3 publications

Bilateral Ocular Myositis Associated with Whipple's Disease

Bilateral Ocular Myositis Associated with Whipple's Disease

Ophthalmic manifestations of Whipple's disease

Ocular Myositis

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