Two Cases of Currarino Syndrome With Improvement of Defecation Function by Dilated Colon Resection

Abstract

We experienced treating two cases of Currarino syndrome in which defecation function was improved by dilated large bowel resection. Their constipation was associated with intestinal dilation following anorectal malformation surgery. Case 1: A boy aged four years and five months was diagnosed as having Currarino syndrome with rectal atresia, sacrococcygeal partial defect, and presacral meningocele as its components. Rectal anastomosis and meningocele resection were performed at the age of 4 months after colostomy, and the colostomy was closed at 7 months of age. An enema, laxative, and herbal medicine were used, but the dilation from the rectum to the sigmoid colon became severe. It was difficult to control defecation, and the expanded intestinal tract was resected. As of 6 years of age, he uses only laxative and herbal medicine. Case 2: A boy aged four years and three months was diagnosed as having Currarino syndrome with anal atresia, sacral malformation, and sacral lipoma as its components. After a colostomy, he underwent anoplasty at six months of age, and the colostomy was closed after one year and one month. He needed an enema and an intestinal lavage after surgery, and the expansion from the rectum to the sigmoid colon became worse, so the dilated intestinal tract was resected. At the age of 12, he has self-defecation but sometimes uses an enema. Hypoperistalsis due to chronic intestinal dilation associated with anorectal malformation was considered to be the cause of constipation.