© Springer Nature Switzerland AG 2021
C. S. Foster et al. (eds.)Uveitishttps://doi.org/10.1007/978-3-030-52974-1_32

32. Whipple’s Disease

Yael Sharon1, 2 and David S. Chu2, 1  
(1)
Metropolitan Eye Research and Surgery Institute, Palisades Park, NJ, USA
(2)
Department of Ophthalmology, Rabin Medical Center, Petah-Tikva, Israel
 
Keywords
UveitisWhipple’s disease

Overview

  • Definition
    • A chronic bacterial infection caused by Tropheryma whipplei

    • Primarily affects the gastrointestinal (GI) tract but may affect other organs, such as lungs, joints, heart, kidney, central nervous system (CNS), and eyes

    • Severe abdominal disease leads to malabsorption and is associated with CNS co-morbidities, and can be potentially fatal

    • Ocular involvement (mainly keratitis, uveitis, and neuro-ophthalmic): 6–8%

    • Well-known yet unexplained propensity in middle-aged, white males

  • Symptoms
    • Blurry vision

    • Floaters

  • Laterality
    • Unilateral or bilateral

  • Course
    • Systemic disease is chronic and relapsing.

    • Ocular disease presents late.

  • Age of onset
    • 55 years

  • Gender/race
    • M:F = 3:1

    • Caucasian

  • Systemic association
    • GI and joint symptoms are most common
      • Weight loss (80–90%)

      • Abdominal pain (50–95%)

      • Diarrhea/steatorrhea (70–85%)

      • Migratory, non-deforming polyarthralgia (70–90%)
        • May precede GI symptoms by months to years

    • Other findings
      • Intermittent, low-grade fever

      • Lymphadenopathy

      • Cardiac (endocarditis, pericarditis, Congestive heart failure (CHF))

      • Pulmonary (pleural effusion, pulmonary infiltrates)

      • CNS (dementia, supranuclear ophthalmoplegia, myoclonus, hypothalamic signs)

    • Possible association with HLA-B27, HLA-DRβ1*13, and DQβ1*06

Exam: Ocular

Anterior Segment

  • Keratitis

  • Mutton-fat keratic precipitates (KPs)

  • Iris nodules

Posterior Segment

  • Vitritis

  • Pars plana snowbanks and snowballs

  • Cotton-wool spots and retinal hemorrhages

  • Vitreous hemorrhage

  • Retinitis and choroiditis

  • Retrobulbar optic neuritis

Neuro-ophthalmic (Less Common than Ocular, about 10%)

  • Gaze palsy

  • Nystagmus

  • Myoclonus

  • Oculomasticatory myorhythmia (OMM)
    • Pathognomonic.

    • Pendular vergent oscillations or smooth vergent nystagmus associated with tongue and mandibular myoclonus.

    • Patients often have gaze paralysis, hypersomnia, and arthralgia without magnetic resonance imaging (MRI) change or GI findings initially.

Exam: Systemic

  • Joint swelling and tenderness (more commonly large joints)

  • Distended abdomen

  • Skin hyperpigmentation and nodules

  • Murmurs (pericardial effusion)

  • Dullness to chest percussion, diminished breath sounds or pleural rub by stethoscope (pleural effusion)

  • Peripheral edema (protein-wasting enteropathy)

  • Altered mental status (confusion, memory loss)

Imaging

  • n/a

Laboratory and Radiographic Testing

  • Gastroscopy with small bowel biopsy is the diagnostic procedure of choice.
    • Light microscopy (LM): clubbed villi and a lamina propria infiltrated with Pas-positive inclusions within and outside of foamy macrophages

    • Electron microscopy (EM): characteristic trilaminar outer cell wall structure (“bacillary bodies”)

    • polymerase chain reaction (PCR) (done also on vitreous sample): high sensitivity; based on the nucleotide sequence of T. whipplei 16S ribosomal RNA

Differential Diagnosis

  • Sarcoidosis

  • Behcet’s disease

  • Collagen vascular diseases

  • Amyloidosis

  • Lyme disease

  • Mycobacterium avium complex (MAC) infection

  • Tuberculosis

  • Histoplasmosis

  • Intraocular lymphoma

Treatment

  • Initial treatment for CNS and ocular involvements
    • Intravenous (IV) ceftriaxone 2 g BID=twice daily plus streptomycin 1 g QD × 2 weeks, or

    • IV trimethoprim sulfamethoxazole (TMP-SMX) 960 mg BID × 1–2 weeks, or

    • IV penicillin, 1.2 million units QD=once daily plus streptomycin 1 g QD × 2 weeks

  • Maintenance therapy for 1–2 years (<1 year is associated with high relapse rate)
    • PO=per os=orally TMP-SMX, 960 mg twice daily +/− rifampin 600 mg QD, or

    • PO doxycycline plus hydroxychloroquine

  • The most common and serious complication is neurologic relapse, even after apparently successful treatment and systemic improvement.

  • Therapeutic and diagnostic vitrectomy for marked vitreous opacities.

Referral/Comanagement

  • Infectious disease

  • Gastroenterology

  • Neurology