만성변비를 가진 뇌이랑없음증환아에서 맹장꼬임 증례1

만성변비를 가진 뇌이랑없음증환아에서 맹장꼬임 증례1

A case of cecal volvulus presenting with chronic constipation in lissencephaly

(지상발표):
Release Date :
Eun-Kyung, Lee, Ji Eun, Kim, Yun-young,Lee, Saeyoon, Kim, Kwang-hae, Choi
Yeungnam University College of Medicine, Daegu, Korea
이은경,김지은,이윤영,김세윤,최광해
영남대의대소아과

Abstract

Background: Cecal volvulus is uncommon in pediatric patients and there are few reports of cecal volvulus with cerebral palsy. Here, we report the case of a patient with a history of mental disability presenting with bowel obstruction diagnosed as cecal volvulus. Case: 19-year-old male patient visited our hospital who presented with abdominal distension, a history of cerebral palsy, refractory epilepsy due to lissencephaly, and chronic constipation. The patient, suffered from chronic constipation over 3yrs, had history of ≤ 2 bowel movements per week with hard large stool, necessitating rectal stimulation for defecation. Abdominal examination showed epigastric and lower abdominal distension with decreased bowel sounds, which had mild rigidity. Laboratory findings showed leukocytosis with a white blood cell count of 45690/μl, increased C-reactive protein at 15.14 mg/dl (reference range 0.5 mg/dl), and increased blood urea nitrogen/creatinine at 33.15/1.91 mg/dl. An abdominal x-ray and computed tomography without contrast enhancement showed fixed dilated bowel intensity in the right lower abdomen. Despite decompression with gastric and rectal tube insertion with antibiotics medications, symptoms did not improved. The patient underwent an exploratory laparotomy that revealed cecal volvulus.. Conclusion : We suggest that cecal and proximal large bowel volvulus should be considered in patients presenting with progressive abdominal distension combined with a history of neuro-developmental delay and constipation.

Keywords: cecal volvulus, lissencephaly,