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一名成年患者出现巨大的完全孤立性重复囊肿,伴有扭转,囊壁由3种不同的黏膜上皮成分构成:病例报告。

A huge completely isolated duplication cyst complicated by torsion and lined by 3 different mucosal epithelial components in an adult: A case report.

作者信息

Xiao-Ming Ai, Jin-Jing Lu, Li-Chen Ho, Lu-Lu Han, Xiong Yue, Hong-Hai Zhang, Nian-Yin Yang

机构信息

Department of General Surgery, BenQ Medical Center, The Affiliated BenQ Hospital of Nanjing Medical University, Nanjing, Jiangsu, China.

出版信息

Medicine (Baltimore). 2018 Nov;97(44):e13005. doi: 10.1097/MD.0000000000013005.

Abstract

RATIONALE

Intestinal or enteric duplication (ED) does exit as a rare congenital malformation of the gastrointestinal system clinically. It is a separate entity, but can be communicated with the gastrointestinal tract. It is characterized by a well-developed muscular wall and lumen endowed with ectopic mucosa, simulating a portion of normal bowel. A completely isolated duplication cyst (CIDC) refers to an extremely uncommon variant of ED, which is secluded from the alimentary tract and possesses its own exclusive blood supply. Surgical procedure is the treatment of choice, because most often, a definitive diagnosis can only be confirmed intraoperatively.

PATIENT CONCERNS

A 20-year-old male patient presented with a 10-day history of intermittent episodes of abdominal pain. The pain evolved from dull into progressive and intolerable, accompanied by vomiting, nausea, and abdominal distention.

DIAGNOSES

Closed-loop small-bowel obstruction with volvulus.

INTERVENTIONS

The patient underwent an emergency exploratory laparotomy.

OUTCOMES

A huge CIDC was observed upon operation, which was affixed to the mesentery with only a narrow base, just like a pedicle; 720° counterclockwise twisting around its base was definitely noted, provoking the compromised blood supply. Complete excision of the cyst was performed along its base safely without violating the intestinal tract. Furthermore, the ectopic mucosa of the cyst exhibited 3 different epithelial lining components histopathologically.

LESSONS

Clinicians should be aware of the possibility of the existence of a duplication and raise a high index of suspicion in case of equivocal diagnosis, particularly in adult population. A low threshold for surgical management should be recommended in order to prevent lethal outcomes.

摘要

原理

肠重复畸形(ED)在临床上确实是一种罕见的胃肠道先天性畸形。它是一个独立的实体,但可与胃肠道相通。其特征是具有发育良好的肌层和含有异位黏膜的管腔,类似正常肠管的一部分。完全孤立性重复囊肿(CIDC)是指ED的一种极其罕见的变异类型,它与消化道隔绝且有自身独立的血供。手术是首选的治疗方法,因为大多数情况下,只有在术中才能确诊。

患者情况

一名20岁男性患者,有10天间歇性腹痛病史。疼痛从隐痛发展为进行性且难以忍受,伴有呕吐、恶心和腹胀。

诊断

闭袢性小肠梗阻伴肠扭转。

干预措施

患者接受了急诊剖腹探查术。

结果

术中发现一个巨大的CIDC,它仅以狭窄的基底附着于肠系膜,宛如一个蒂;明确观察到其围绕基底逆时针扭转720°,导致血供受损。沿囊肿基底安全地完整切除囊肿,未侵犯肠道。此外,囊肿的异位黏膜在组织病理学上表现出3种不同的上皮内衬成分。

经验教训

临床医生应意识到存在重复畸形的可能性,在诊断不明确时,尤其是在成年患者中,要提高警惕。应建议采取较低的手术治疗阈值,以防止出现致命后果。

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