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

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
• posterior ocular involvement
• retinal imaging
• optic disc edema

Acknowledgements

We thank Anna Beltrame, MD, from the Centre for Tropical disease of Sacro Cuore Don Calabria Hospital of Negrar for the systemic management of the patient and Giulia Querzoli, MD, from the Pathology Unit of Sacro Cuore Don Calabria Hospital of Negrar for the histopathology evaluation.