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Congenital multiple colonic atresias with intestinal malrotation: a case report.

ABSTRACT: BACKGROUND:Congenital intestinal atresia develops in 1 in 1500 to 20,000 births. Colonic atresia, which accounts for 1.8-15% of intestinal atresia cases, is accompanied by other gastrointestinal atresias such as small intestinal atresia, gastroschisis, imperforate anus, and intestinal malformation in 47-80%. Although a report shows that patients with multiple colonic atresias are 8.9% of those with colonic atresia. CASE PRESENTATION:A male infant did not have the first bowel movement within 36?h of birth and had abdominal distention/vomiting. Radiography showed significant dilation of the intestinal tract. A contrast enema examination at 3 days of age showed a microcolon and disruption in the descending colon. We performed an emergency decompressive loop enterostomy in the distended segment. At the age of 7 months, imaging from the stoma showed disruption of the contrast medium in the intestinal tract at the right lower abdomen, and the continuity of the intestinal tract was not clarified. Intestinal malrotation was found during the second surgery, and the enterostomy was located in the ileum proximal to Bauhin's valve. Continuity of the intestinal serosal surface was maintained. However, multiple membranous obstructions (three atresias and one stenosis) were observed in the distal segment of the bowel, which was penetrated by intraluminal advancement of a urethral catheter. Therefore, he was diagnosed with multiple colonic atresias. The intestinal tract was longitudinally incised, and membranectomy and mucosal/lateral suture were performed. CONCLUSIONS:It is important for neonates with intestinal atresia to evaluate and prepare for distal patency of the colon before radical anastomosis. In addition, anomalies associated with colon atresia should also be assessed.

SUBMITTER: Ishii D

PROVIDER: S-EPMC7105550 | biostudies-literature | 2020 Mar

REPOSITORIES: biostudies-literature

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Congenital multiple colonic atresias with intestinal malrotation: a case report.

Ishii Daisuke D Miyagi Hisayuki H Hirasawa Masatoshi M Miyamoto Kazutoshi K

Surgical case reports 20200330 1

<h4>Background</h4>Congenital intestinal atresia develops in 1 in 1500 to 20,000 births. Colonic atresia, which accounts for 1.8-15% of intestinal atresia cases, is accompanied by other gastrointestinal atresias such as small intestinal atresia, gastroschisis, imperforate anus, and intestinal malformation in 47-80%. Although a report shows that patients with multiple colonic atresias are 8.9% of those with colonic atresia.<h4>Case presentation</h4>A male infant did not have the first bowel movem ...[more]

PMID: 32232592

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Project description:Introductionand importance: Caecal volvulus represents 30% of colonic volvulus. It happens due to torsion or hyperflexion of a hypermobile caecum. Usually it is secondary to an axial rotation of the caecum and the ileum around the mesentery. On the other hand Intestinal malrotation occurs due to incomplete or faulty rotation and fixation of the gut during fetal life. The occurrence of these two anomalies together is scarse which makes this case report interesting.Case presentationA 75 year old man with medical history of terminal kidney failure, presented to the emergency room with an intestinal obstruction syndrome. On examination the patient had a distended abdomen with tenderness in the left upper quadrant. Biology found an important biological inflammatory syndrome with hyperleukocytosis and elevated CRP. Plain X-ray of the abdomen in erect posture showed an air fluid colonic level in the left hypochondrium. CT scan showed signs of caecal volvulus with intestinal malrotation. A brief reanimation and nasogastric aspiration couldn't solve the problem therefore emergency laparotomy was needed ileocaecal resection was performed associated with LADD's procedure in order to treat both anomalies and prevent further gut volvulus.Clinical discussionDespite it's rareness, caecul volvulus represents the second cause of large bowel volvulus just behind sigmoid volvulus. Intestinal malrotation in adults subjects is estimated to occur in 0.2-0.5%.The uniqueness of our case is that these two anomalies were associated in such a way that it made both the diagnosis and the therapy even more difficult. Abdominal CT has become mandatory for pre-operative diagnosis of intestinal volvulus. Surgery is the gold standard treatment for caecal volvulus. The usual options are manual detorsion, carcopexy, caecostomy and colectomy.ConclusionThis case reports a rare association of a caecum volvulus with intestinal malrotation that emphasis the place of modern technologies such as CT scan in order to achieve correct preoperative diagnosis. We also describe our approach to this uncommon surgical emergency in order to provide an efficient treatement.

Dataset Information

Congenital multiple colonic atresias with intestinal malrotation: a case report.

Publications

Congenital multiple colonic atresias with intestinal malrotation: a case report.

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