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一例罕见的小儿病例:肠道重复畸形酷似梅克尔憩室扭转的异常表现

An unusual presentation of intestinal duplication mimicking torsion of Meckel's diverticulum: a rare report of a pediatric case.

作者信息

Matsukubo Makoto, Muto Mitsuru, Kedoin Chihiro, Matsui Mayu, Murakami Masakazu, Sugita Koshiro, Yano Keisuke, Onishi Shun, Harumatsu Toshio, Yamada Koji, Yamada Waka, Kaji Tatsuru, Ieiri Satoshi

机构信息

Department of Pediatric Surgery, Research Field in Medical and Health Sciences, Medical and Dental Area, Research and Education Assembly, Kagoshima University, 8-35-1, Sakuragaoka, Kagoshima, 8908520, Japan.

Clinical Training Center, Kagoshima University Hospital, Kagoshima, Japan.

出版信息

Surg Case Rep. 2022 Mar 28;8(1):53. doi: 10.1186/s40792-022-01409-6.

Abstract

BACKGROUND

Enteric duplication is a congenital disease that occurs throughout the entire gastrointestinal tract. Although it may sometimes cause intestinal volvulus, a few reports have described cases of enteric duplication twisted on itself. We experienced a rare pediatric case of long-segment tubular ileal duplication showing torsion. Torsion of enteric duplication is extremely rare. We herein report a pediatric case showing unusual torsion of ileal duplication requiring emergency surgery.

CASE PRESENTATION

A 3-year-old boy presented with abdominal pain and vomiting. Contrast-enhanced computed tomography (CT) revealed a cystic luminal structure with a blind end and fluid collection in the pelvic cavity. CT also showed no findings of ileus or intestinal dilatation except for a cystic luminal structure. The preoperative diagnosis was torsion of Meckel's diverticulum. The patient underwent emergent explorative diagnostic laparoscopy. As a result, a necrotic luminal structure and bloody ascites were recognized, and small-scale laparotomy was performed. Long-segment ileal duplication was recognized. The long-segment tubular ileal duplication shared the anti-mesenteric side of the intestinal wall along one-third of its length. The residual two-thirds of its length was free from the ileum and its blind end was twisted in a manner that looked similar to Meckel's diverticulum. Normal ileum and the duplication, including the twisted necrotic portion, were resected, and ileal anastomosis was performed. The postoperative course was uneventful. A pathological examination confirmed the definitive diagnosis of enteric duplication.

CONCLUSIONS

We reported the unusual presentation of intestinal duplication mimicking torsion of Meckel's diverticulum. Enteric duplication shows various clinical symptoms and presentations. We must understand that the classification of digestive enteric duplication is diverse with a variety of associated clinical symptoms.

摘要

背景

肠重复畸形是一种发生于整个胃肠道的先天性疾病。尽管它有时可能导致肠扭转,但仅有少数报告描述过肠重复畸形自身发生扭转的病例。我们遇到了一例罕见的小儿长段管状回肠重复畸形发生扭转的病例。肠重复畸形扭转极为罕见。在此,我们报告一例小儿回肠重复畸形发生异常扭转并需要急诊手术的病例。

病例报告

一名3岁男孩出现腹痛和呕吐症状。增强计算机断层扫描(CT)显示盆腔内有一个盲端的囊性管腔结构及积液。CT还显示除了囊性管腔结构外,没有肠梗阻或肠扩张的表现。术前诊断为梅克尔憩室扭转。患者接受了急诊探查诊断性腹腔镜检查。结果,发现了一个坏死的管腔结构和血性腹水,随后进行了小切口剖腹手术。术中发现了长段回肠重复畸形。长段管状回肠重复畸形在其长度的三分之一处沿肠壁的系膜对侧走行。其剩余三分之二的长度与回肠分离,且盲端扭转,外观类似梅克尔憩室。切除了正常回肠及包括扭转坏死部分的重复畸形,进行了回肠吻合术。术后病程顺利。病理检查确诊为肠重复畸形。

结论

我们报告了一例模仿梅克尔憩室扭转的肠重复畸形的异常表现。肠重复畸形表现出各种临床症状和表现。我们必须认识到消化性肠重复畸形的分类多样,伴有各种相关的临床症状。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/cad7/8960505/b5a782d51a44/40792_2022_1409_Fig1_HTML.jpg

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