Recurrent orbital inflammation and Whipple disease

Abstract

Purpose: To describe a recurrent case of idiopathic orbital inflammation (OI) associated with Whipple disease.

Methods: Observational case report.

Results: A 63-year-old woman developed myositis or idiopathic orbital inflammation (OI). Whipple disease had been diagnosed 1 year before by duodenal biopsy. OI appeared 1 year following the end of a long course of trimethoprim-sulfamethoxazole treatment. During follow-up months later, recurrent OI was noted in the same and contralateral orbits. Flare-ups of OI were suggestive of disease relapse and subsequent duodenal biopsy confirmed the presence of PAS-positive particles in macrophages. Treatment with trimethoprim-sulfamethoxazole and corticosteroids was necessary to prevent recurrences.

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