Intest Res.  2021 Jan;19(1):119-125. 10.5217/ir.2019.09177.

Whipple disease mimicking inflammatory bowel disease

• Tatsuki M
• Ishige T
• Igarashi Y
• Hatori R
• Hokama A
• Hirato J
• Muise A
• Takizawa T
• Arakawa H

• KMID: 2512108
• DOI: http://doi.org/10.5217/ir.2019.09177

Abstract

Whipple disease is a systemic chronic infection caused by Tropheryma whipplei. Although chronic diarrhea is a common gastrointestinal symptom, diagnosis is often difficult because there are no specific endoscopic findings, and the pathogen is not detectable by stool culture. We present a female patient with Whipple disease who developed chronic bloody diarrhea and growth retardation at the age of 4 years. Colonoscopy showed a mildly edematous terminal ileum and marked erythema without vascular patterns throughout the sigmoid colon and rectum. Subsequently, a primary diagnosis of ulcerative colitis was made. Histopathological analysis of the terminal ileum showed the presence of foamy macrophages filled with periodic acidSchiff-positive particles. Polymerase chain reaction using DNA from a terminal ileum biopsy sample amplified a fragment of 16S rRNA from T. whipplei. Antibiotic treatment relieved the patient’s symptoms. There was no evidence of immunodeficiency in the present case. Since Whipple disease worsens after anti-tumor necrosis factor inhibitor therapy, considering this infection in the differential diagnosis may be important in patients with inflammatory bowel disease, especially before initiation of immunotherapy.

Keyword

Colitis, ulcerative; Child, preschool

Figure

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  Fig. 1. Endoscopic image of the terminal ileum (A) and sigmoid colon (B) of the patient at the time of admission. The ileum showed mild edema. Colonoscopy showed friability, erythema, absence of vascular patterns, and erosions.

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  Fig. 2. Histological image of the biopsy specimens from the duodenum and terminal ileum revealed foamy macrophages with intracellular granules. Histological image of the terminal ileum with foamy macrophages. (A) Foamy macrophages are evident in the sample with H&E staining (arrow, ×100). (B) Periodic acid-Schiff positive macrophages are seen in the lamina propria (arrows, ×400).

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  Fig. 3. Electron microscopic findings of the biopsy sample and polymerase chain reaction (PCR) detection of Tropheryma whipplei from the terminal ileum. (A) Numerous rod-shaped bacilli are observed (arrows). (B) The lane shows a PCR product of 160 base pairs specific to the T. whipplei 16S ribosomal RNA.

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  Fig. 4. Endoscopic findings after 12 months of treatment for Whipple disease. Duodenal edema is improved (A), as well as ileal (B) and colonic (C, D) pathological findings.

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