Recurrent orbital inflammation and Whipple disease

Purpose: To describe a recurrent case of idiopathic orbital inflammation (01) associated with Whipple disease. Methods: Observational case report. Results: A 63-year-old woman developed myositis or idiopathic orbital inflammation (01). Whipple disease had been diagnosed I year before by duodenal biopsy. 01 appeared 1 year following the end of a long course of trimethoprim-sulfamethoxazole treatment. During follow-up months later, recurrent 01 was noted in the same and contralateral orbits. Flare-ups of 01 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 01 picture that is immmunologically mediated.