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A Case of Enteritis Cystica Profunda Accompanied by a Lipoma in the Duodenal Bulb

십이지장 구부에 발생한 심재성 낭종성 장염에서 지방종을 동반한 1예

  • Lee, Dae Sung (Department of Internal Medicine, Hanyang University College of Medicine) ;
  • Jeong, Hae Ryong (Department of Internal Medicine, Hanyang University College of Medicine) ;
  • Kim, Jin Ok (Department of Internal Medicine, Hanyang University College of Medicine) ;
  • Tae, Hye Jin (Department of Internal Medicine, Hanyang University College of Medicine) ;
  • Choi, Ho Soon (Department of Internal Medicine, Hanyang University College of Medicine) ;
  • Ahn, Hye In (Department of Pathology, Hanyang University College of Medicine) ;
  • Paik, Seung Sam (Department of Pathology, Hanyang University College of Medicine)
  • 이대성 (한양대학교 의과대학 내과학교실) ;
  • 정해룡 (한양대학교 의과대학 내과학교실) ;
  • 김진옥 (한양대학교 의과대학 내과학교실) ;
  • 태혜진 (한양대학교 의과대학 내과학교실) ;
  • 최호순 (한양대학교 의과대학 내과학교실) ;
  • 안혜인 (한양대학교 의과대학 병리학교실) ;
  • 백승삼 (한양대학교 의과대학 병리학교실)
  • Received : 2012.07.29
  • Accepted : 2012.09.24
  • Published : 2014.03.01

Abstract

Enteritis cystica profunda (ECP) is a very rare hyperplastic benign lesion of the small intestine. Histologically, it is characterized by mucin-filled cystic spaces frequently lined by benign epithelium within the submucosa and muscularis propria. ECP occurs much less frequently than gastritis cystica profunda (GCP) or colitis cystica profunda (CCP). It is most often seen in adults with Crohn's disease, Peutz-Jeghers syndrome, hamartomatous polyp, or congenital anomalies. We encountered a case of ECP ($90{\times}15 mm$) accompanied by an elongated lipoma in the duodenal bulb, in the absence of any other disease. An endoscopic polypectomy proved sufficient for diagnosing and treating the ECP. To our knowledge, this is the first reported case of ECP accompanied by a lipoma in the duodenal bulb in the absence of any other intestinal disease.

ECP는 드문 양성 질환으로 알려져 있으며 대부분 다른 동반 질환이 있는 경우 발견된다. 저자들이 경험한 십이지장구부에 길이 90 mm의 기다란 관상구조의 폴립모양 병변에서 확인된 ECP는 지방종을 동반한 예로 아직까지 국내외에서 증례를 찾아볼 수 없었다. 유사한 예로 국외에서 1예가 보고되었지만 본 증례와 차이점은 회장에서 발생하였고 장중첩증의 합병증을 동반한 경우였다. 저자들은 기저 질환 및 증상이 없는 환자에서 우연히 발견한 십이지장구부의 기다란 폴립모양 병변의 ECP에서 지방종을 동반한 예를 경험하였기에 이를 보고한다.

Keywords

References

  1. Jeong HK, Kim JK, Hwang IS, et al. A case of enteritis cystica profunda in the duodenum. Korean J Gastroenterol 2002;39:55-58.
  2. You KW, Park SW, Lee GS, Kim DJ, Moon HC, Hong GY. A case of enteritis cystica profunda in the ampulla of vater mimicking choledochocele. Clin Endosc 2013;46:178-181. https://doi.org/10.5946/ce.2013.46.2.178
  3. Kim IG, Kim CH, Kim CG, et al. A case of Peutz-Jeghers syndrome with small bowel perforation and enteritis cystica profunda. Korean J Gastroenterol 1997;29:677-682.
  4. Chang HJ, Jung JI, Kim MC, et al. A case of enteritis cystica profunda in the duodenal bulb. Korean J Gastrointest Endosc 2005;31:419-422.
  5. Goodall HB, Sinclair ISR. Colitis cystica profunda. J Pathol Bacteriol 1957;73:33-42. https://doi.org/10.1002/path.1700730106
  6. Pan YS, Kuo HT, Lai HY, Chuang SS, Liu CS. Solitary ileal lipoma presenting with ileocolic intussusception: an unusual cause of enteritis cystica profunda. J Formos Med Assoc 1997;96:469-472.
  7. Sunagawa H, Kinjyou T, Nagahama M, Nishimaki T, Nakayama T. Enteritis cystica profunda presenting as ileoileal intussusception in a child: report of a case. Surg Today 2005;35:164-167. https://doi.org/10.1007/s00595-004-2912-4
  8. Kyriakos M, Condon SC. Enteritis cystica profunda. Am J Clin Pathol 1978;69:77-85. https://doi.org/10.1093/ajcp/69.1.77
  9. Guest CB, Reznick RK. Colitis cystica profunda: review of the literature. Dis Colon Rectum 1989;32:983-988. https://doi.org/10.1007/BF02552279
  10. Iwanaga T, Koyama H, Takahashi Y, Taniguchi H, Wada A. Diffuse submucosal cysts and carcinoma of the stomach. Cancer 1975;36:606-614. https://doi.org/10.1002/1097-0142(197508)36:2<606::AID-CNCR2820360243>3.0.CO;2-V