Department of Surgery, Seoul St. Mary's Hospital, The Catholic University of Korea, Seoul, Korea.
Medicine (Baltimore). 2021 May 7;100(18):e25772. doi: 10.1097/MD.0000000000025772.
Gastrointestinal tract duplication is a rare congenial anomaly which can be found anywhere along the gastrointestinal tract. While many patients are incidentally diagnosed during operation, in some cases it can present with severe gastrointestinal symptoms. In this case report, the patient presented with signs of toxic megacolon leading to rapid aggravation of inflammatory shock.
A 49-day old male infant presented with fever, poor feeding, and severe abdominal distension.
Abdominal ultrasonography was done. During the examination, a foley catheter was inserted through the anus to evaluate bowel patency and enable rectal decompression. The tip of the foley catheter was located in a separate narrower tubular lumen adjacent to the distended rectum. These findings suggested possibility of a tubular duplication cyst of the rectum as the culprit for the bowel obstruction.
The patient underwent emergency laparotomy. Findings showed multiple tubular intestinal duplications involving the ileum, appendix, cecum, descending colon, sigmoid colon and rectum. The true lumen of the rectosigmoid colon was completely collapsed while the adjacent tubular cyst remained severely distended and stool passage was not possible. Decompression of the sigmoid colon was done with loop colostomy with both the wall of the true bowel and enteric cyst forming the colostomy orifice.
After 40 days of postoperative care, the patient was discharged with no immediate complications. Four months after the initial operation, colostomy take-down and transanal rectal common wall division was done. No complications were observed.
To our knowledge, this is the first case to be reported where a rare presentation of intestinal duplication resulted in an acute presentation toxic megacolon. Such emergency cases can be effectively treated with emergency surgical bowel decompression and elective common wall division.
胃肠道重复畸形是一种罕见的先天性异常,可发生于胃肠道的任何部位。虽然许多患者在手术中偶然被诊断出来,但在某些情况下,它可能会出现严重的胃肠道症状。在本病例报告中,患者表现出中毒性巨结肠的迹象,导致炎症性休克迅速恶化。
一名 49 天大的男婴出现发热、喂养不良和严重腹胀。
进行了腹部超声检查。在检查过程中,通过肛门插入 Foley 导管以评估肠道通畅性并进行直肠减压。Foley 导管尖端位于与扩张直肠相邻的单独较窄的管状腔室内。这些发现提示可能存在直肠管状重复囊肿引起的肠梗阻。
患者接受了紧急剖腹手术。检查结果显示多个管状肠重复畸形累及回肠、阑尾、盲肠、降结肠、乙状结肠和直肠。直肠乙状结肠的真腔完全塌陷,而相邻的管状囊肿仍然严重扩张,无法排便。通过乙状结肠造口术对乙状结肠进行减压,真肠壁和肠囊肿形成造口口。
术后 40 天的护理后,患者无立即并发症出院。初次手术后 4 个月,进行了造口关闭和经肛门直肠共同壁分割术。未观察到并发症。
据我们所知,这是首例报告肠道重复畸形罕见表现导致急性中毒性巨结肠的病例。此类紧急情况可通过紧急手术肠道减压和选择性共同壁分割有效治疗。