Central Nervous System Relapse of Whipple's Disease

Kiyoaki Uryu; Takashi Sakai; Tomonori Yamamoto; Takahito Mae; Yoshie Iwasaki Willard; Hiroji Sugita; Koukichi Asano; Kenichi Yamashita; Hiromasa Harada; Kou Fukuda

doi:10.2169/internalmedicine.51.7304

CASE REPORTS

Central Nervous System Relapse of Whipple's Disease

Kiyoaki Uryu, Takashi Sakai, Tomonori Yamamoto, Takahito Mae, Yoshie Iwasaki Willard, Hiroji Sugita, Koukichi Asano, Kenichi Yamashita, Hiromasa Harada, Kou Fukuda

Author information

Kiyoaki Uryu
Department of Respiratory Medicine, Yao Tokushukai General Hospital, Japan
Takashi Sakai
Department of General Internal Medicine, Obihiro Daiichi Hospital, Japan
Tomonori Yamamoto
Department of Gastroenterology, Yao Tokushukai General Hospital, Japan
Takahito Mae
Department of Respiratory Medicine, Yao Tokushukai General Hospital, Japan
Yoshie Iwasaki Willard
Department of Cytology, Yao Tokushukai General Hospital, Japan
Hiroji Sugita
Department of Gastroenterology, Yao Tokushukai General Hospital, Japan
Koukichi Asano
Department of Gastroenterology, Yao Tokushukai General Hospital, Japan
Kenichi Yamashita
Department of Pathology, Yao Tokushukai General Hospital, Japan
Hiromasa Harada
Department of Respiratory Medicine, Yao Tokushukai General Hospital, Japan
Kou Fukuda
Department of Cardiovascular Medicine, Yao Tokushukai General Hospital, Japan

Keywords: Whipple's disease, central nervous system disease, polymerase chain reaction, CTRX; ceftriaxone sodium, TMP-SMX; trimethoprim-sulfamethoxazole

JOURNAL OPEN ACCESS

2012 Volume 51 Issue 15 Pages 2045-2050

DOI https://doi.org/10.2169/internalmedicine.51.7304

Details

Abstract

A 50-year-old man presented with a 12 kg weight loss in 8 months. Upper gastrointestinal endoscopy findings showed strong erosion and diffuse bleeding in the duodenum. Histopathological findings showed PAS staining-positive macrophages consistent with Whipple's disease. He was treated with trimethoprim-sulfamethoxazole. His condition initially improved. However, during his 6-year course of treatment he developed a central nervous system relapse. Tropheryma whipplei DNA was detected by a polymerase chain reaction in his cerebrospinal fluid. This relapse was successfully treated with ceftriaxone sodium (CTRX). We considered that as initial therapy for Whipple's disease, it would be important to administer CTRX for at least a few months, due to its high translatability to CSF.

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