Whipple\u2019s disease scleral nodules: a novel presentation in 2 consecutive patients

Waleed K Alsarhani,Azza M Y Maktabi,Hind M Alkatan,Afaf L Alsolami,Muhannad I Alkhalifah,Adel H Alsuhaibani

doi:10.1186/s12886-020-01695-4

Waleed K Alsarhani, Azza M Y Maktabi + Show 4 more

Open Access

https://doi.org/10.1186/s12886-020-01695-4

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Abstract

BackgroundWhipple’s disease (WD) is a rare, chronic, infection caused by gram-positive filamentous aerobic actinobacterium Tropheryma whipplei occurs classically in the gastrointestinal tract and shows histopathologically foamy macrophages with typical numerous PAS-positive, non-acid fast particles. Ocular WD in the form of uveitis may occur in the absence of systemic disease but has not been reported to present with scleral manifestation. We describe for the first time to the best of our knowledge 2 cases of scleral nodules with typical histopathological morphology of WD and without systemic involvement.Case presentationThe first was a 53-year old diabetic male farmer who presented with 2 nontender right eye scleral nodules for 3 months, had a negative systemic workup, and surgical excision showed Periodic acid Schiff (PAS)-positive eosinophilic structures inside macrophages. Grocott’s methenamine silver (GMS) stain and acid-fast bacilli (AFB) stain of the tissue itself were negative. The second case was a 60-year old male who presented with an asymptomatic superior scleral nodule for 4 months, which showed similar appearance and negative GMS and AFB stains.ConclusionWD should be included in the differential diagnosis of scleral nodules even in the absence of systemic symptoms. Surgical excision without systemic treatment resulted in successful outcome without recurrence.

Highlights

Whipple’s disease (WD) is a rare, chronic, infection caused by gram-positive filamentous aerobic actinobacterium Tropheryma whipplei occurs classically in the gastrointestinal tract and shows histopathologically foamy macrophages with typical numerous Periodic acid Schiff (PAS)-positive, non-acid fast particles
WD should be included in the differential diagnosis of scleral nodules even in the absence of systemic symptoms
Surgical excision without systemic treatment resulted in successful outcome without recurrence

Summary

Introduction

Whipple’s disease (WD) is a rare, chronic, infection caused by gram-positive filamentous aerobic actinobacterium Tropheryma whipplei occurs classically in the gastrointestinal tract and shows histopathologically foamy macrophages with typical numerous PAS-positive, non-acid fast particles. Isolated scleral involvement has never been reported in the literature to the best of our knowledge. * Correspondence: hindkatan@yahoo.com; hkatan@ksu.edu.sa 1Ophthalmology Department, College of Medicine, King Saud University, Riyadh, Saudi Arabia 2Pathology Department, College of Medicine, King Saud University Medical city, King Saud University, Riyadh, Saudi Arabia Full list of author information is available at the end of the article report two cases of three painless scleral nodules with histopathologic evidence of WD.

Results

Conclusion