Unique Features of Posterior Ocular Involvement of Whipple's Disease in a Patient with No Gastrointestinal Symptoms

Ocul Immunol Inflamm. 2022 Jul;30(5):1168-1171. doi: 10.1080/09273948.2020.1859548. Epub 2021 Feb 5.

Abstract

Purpose: To describe posterior ocular involvement features of Whipple's disease (WD) in a patient with no gastrointestinal symptoms.

Methods: Retrospective case report.

Observation: A 53-year-old man with a 2-year history of seronegative arthritis presented with bilateral intraocular inflammation, optic disc edema, and cystoid macular edema (CME) in the left eye. A diagnosis of noninfectious uveitis was made and oral prednisolone was started. Despite initial improvement, after 6 weeks, CME was found in both eyes. Because of the initial response, the anti-tumor necrosis factor agent Adalimumab was started. Twelve weeks after initiation of adalimumab, fundus examination revealed widespread dot-blot retinal hemorrhages and multifocal chorioretinal lesions at the posterior pole and mid-periphery. The chorioretinal lesions appeared as hyperreflective drusen-like deposits located in the sub-retinal pigment epithelium (RPE) space on the tomographic scan. WD was considered and confirmed by polymerase chain reaction test and duodenal biopsy.

Conclusion: Posterior ocular involvement in WD may present with a wide clinical spectrum including intraocular inflammation and unique features of sub-RPE deposits, widespread retinal hemorrhages, and optic disc edema.

Keywords: Whipple’s disease; intraocular inflammation; optic disc edema; posterior ocular involvement; retinal imaging.

Publication types

  • Case Reports

MeSH terms

  • Adalimumab / therapeutic use
  • Fluorescein Angiography
  • Humans
  • Inflammation / complications
  • Macular Edema* / diagnosis
  • Macular Edema* / drug therapy
  • Macular Edema* / etiology
  • Male
  • Middle Aged
  • Papilledema* / diagnosis
  • Prednisolone
  • Retinal Hemorrhage
  • Retrospective Studies
  • Uveitis* / complications
  • Whipple Disease* / complications
  • Whipple Disease* / diagnosis
  • Whipple Disease* / drug therapy

Substances

  • Prednisolone
  • Adalimumab