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Long-term Treatment of Chronic Enteropathy Associated with SLCO2A1 Gene(CEAS)Accompanied by Primary Hypertrophic Osteoarthropathy Tsuneyoshi Yao 1 , Makoto Yorioka 1 , Akihiro Koga 1 , Takao Kanemitsu 2,3 , Mikihiko Moriyama 2,4 , Satoshi Ishikawa 2,3 , Fumihito Hirai 2,5 , Junji Umeno 6 , Takayuki Matsumoto 7,8 , Akinori Iwashita 9 1Sada Hospital, Fukuoka, Japan 2ex- Sada Hospital, Fukuoka, Japan 3Department of Endoscopy, Fukuoka University Chikushi Hospital, Chikushino, Japan 4Moriyama Clinic, Saga, Japan 5Inflammatory Bowel Disease Center, Fukuoka University Chikushi Hospital, Chikushino, Japan 6Department of Medicine and Clinical Science, Graduate School of Medical Sciences, Kyushu University, Fukuoka, Japan 7ex- Department of Medicine and Clinical Science, Graduate School of Medical Sciences, Kyushu University, Fukuoka, Japan 8Division of Gastroenterology, Department of Internal Medicine, Faculty of Medicine, Iwate Medical University, Morioka, Japan 9Department of Pathology, Fukuoka University Chikushi Hospital, Chikushino, Japan Keyword: CNSU , CEAS , 肥厚性皮膚骨膜症 , PHO , ヘリカルCT pp.1467-1476
Published Date 2017/10/25
DOI https://doi.org/10.11477/mf.1403201200

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 A 73-year-old male patient developed CEAS(chronic enteropathy associated with SLCO2A1 gene)at age 12, and he had presented with anemia and anemic symptoms. Over the next 19 years, he was frequently hospitalized for treatment, and he received intravenous injections of iron preparations and blood transfusions. At age 29, he underwent gastrectomy because of a gastric ulcer(Helicobacter pylori antibodies:negative). At ages 30 and 31, he underwent ileoileostomy(ileal-ileal anastomosis)and blind loop resection, respectively. At age 65, he underwent EBD(endoscopic balloon dilatation)at the anastomosis site because of repeated ileus symptoms. After 42 years of blind loop resection, he was able to be managed with oral administration of iron preparations. With no anemic symptoms, he was able to engage in moderate-level labor and lead a normal life. Over the 8 years since EBD and till date, he has been symptom-free, and no anastomotic stricture has recurred.

 Here, I report on the clinical progress of a patient with CEAS that followed a favorable course, together with a presentation of resected specimens, X-ray, and endoscopic images, and a discussion of the details. The results also indicate that helical computed tomography was extremely useful for the diagnosis of cutis verticis gyrata accompanying primary hypertrophic osteoarthropathy.


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