Chorioretinitis and vitreitis due to Tropheryma whipplei after transplantation: case report and review

Abstract

Whipple's disease is a very rare chronic multisystemic bacterial disease characterized by diarrhea, malabsorption, fever, and polyarthritis. Ocular manifestations occur very rarely. Previous reports have suggested that the use of immunosuppressive drugs appears to accelerate or exacerbate the clinical course of Whipple's disease; however, the illness has yet to be reported in the setting of transplantation. Herein, we describe what we believe is the first reported case of Whipple's disease after transplantation. The patient is a 51-year-old woman who developed progressive visual floaters and blurring of vision 30 years after living-related kidney transplantation for an autosomal-dominant polycystic kidney disease. Her allograft was functioning well on maintenance immunosuppressive therapy with azathioprine and prednisone when she developed visual abnormalities. Transient weight loss, gastrointestinal symptoms, and migratory polyarthralgia predated the onset of ocular disease by several years. The diagnosis of Whipple's bilateral vitreitis and chorioretinitis was confirmed by polymerase chain reaction analysis demonstrating Tropheryma whipplei nucleic acid in vitreous fluid and peripheral blood sample as well as by demonstration of the bacilli by cytopathology. Intraocular vancomycin, intravenous ceftriaxone, and prolonged course of oral trimethoprim-sulfamethoxazole therapy led to clinical improvement and recovery of visual acuity.

Differential Accumulation of Secreted AβPP Metabolites in Ocular Fluids1

“…Although the collection of ocular fluids raises concern for safety and acceptance, eyes have been safely tapped for a number of ocular treatments (e.g., [35–37]). In addition, there are patents for the diagnosis of AD based on the activity of acetylcholinesterase activity in ocular fluids [38].…”

Renal amyloidosis in Whipple disease: a case report

“…Another important issue is the implementation of immunosuppressive treatment (cyclophosphamide and prednisone) due to renal amyloidosis what probably led to severe relapse of Whipple disease. Such negative association between Whipple disease and immunosuppressive medications was suggested previously by several authors [16-19]. After administration of antibiotic therapy the patient’s general condition improved dramatically, however, kidneys injury was irreversible and the patient required renal replacement therapy.…”

Tropheryma whippleiCrystalline Keratopathy: Report of a Case and Updated Review of the Literature

“…Anterior segment involvement may include keratitis, iritis, iris nodules, and peripheral corneal opacities and neovascularization [20, 21]. Posterior segment findings include pars plana snowbanks, snowballs, chorioretinitis, retinal vasculitis and capillary nonperfusion, cystoid macular edema, branch retinal artery occlusions, cotton wool spots, retinal and vitreous hemorrhages, optic nerve swelling, and choroidal folds [10, 13, 15, 22]. …”

“…PCR from aqueous and vitreous fluid have established the diagnosis in several cases [9, 11, 12, 14, 15]. Vitreous biopsies have demonstrated PAS-positive inclusions in macrophages [4, 10] and nonspecific chronic inflammation [4].…”