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乙状结肠囊性重复畸形的一种罕见表现,其内部完全被鳞状上皮覆盖。

An unusual presentation of a cystic duplication of the sigmoid colon entirely lined with squamous epithelium.

作者信息

Rodesch Gregory, Dargent Jean Louis, Haller Annick, Cassart Marie, Avni Freddy, Cos Theresa, Van Herreweghe Inge, Deleuze Jean François, Lingier Pierre

机构信息

Department of Pediatric Surgery, University Chidren's Hospital Queen Fabiola, Brussels, Belgium.

出版信息

J Pediatr Surg. 2009 Sep;44(9):1831-4. doi: 10.1016/j.jpedsurg.2009.05.029.

DOI:10.1016/j.jpedsurg.2009.05.029
PMID:19735835
Abstract

Alimentary tract duplications are rare congenital malformations that occur most commonly in the jejunoileal part of the gastrointestinal tract. Management of this pathologic condition is usually drawn up. We report a case of descending colonic communicating duplication in which clinical presentation and anatomopathologic results were unexpected. A slightly echogenic abdominal mass reaching 72 x 36 mm in the left flank was diagnosed in a female fetus during the third trimester ultrasound examination. At birth, volume of the mass rapidly evolved, and despite no intestinal obstruction was observed by compression of the adjacent gastrointestinal tract, abdomen was distended. Abdominal plain film showed a large air collection, and the barium enema demonstrated a slight leak of contrast in the aerated mass, suggesting a communication with the sigmoid colon. No other abnormalities were seen. The patient underwent surgery in emergency. The mass was then totally excised through an antimesenteric resection of the tubular tract joining cystic mass and sigmoid colon. A lateral suture of the colon was subsequently performed. The wall of the duplication is usually composed of a smooth muscle layer covered by an epithelium, mostly of intestinal type. Herein, we describe a descending colonic duplication completely lined with nonkeratinizing squamous epithelium. Therefore, the association of a colonic mucosa (of endodermic origin) and a squamous epithelium (derived from the ectoderm) in our case is an interesting finding and is not explained by the various theories. Furthermore, the clinical characteristics, diagnosis, and treatment of intestinal duplications are discussed with regard to literature.

摘要

消化道重复畸形是一种罕见的先天性畸形,最常发生于胃肠道的空肠回肠段。通常会制定针对这种病理状况的治疗方案。我们报告一例降结肠交通性重复畸形病例,其临床表现和解剖病理结果出人意料。在孕晚期超声检查时,一名女胎的左腹侧被诊断出一个大小为72×36毫米、回声略强的腹部肿块。出生时,肿块体积迅速增大,尽管通过压迫相邻胃肠道未观察到肠梗阻,但腹部膨隆。腹部平片显示有大量积气,钡剂灌肠显示充气肿块中有轻微造影剂渗漏,提示与乙状结肠相通。未见其他异常。患者接受了急诊手术。随后通过对连接囊性肿块和乙状结肠的管状结构进行系膜对侧切除,将肿块完全切除。随后对结肠进行了侧方缝合。重复畸形的壁通常由一层平滑肌层覆盖着上皮构成,上皮大多为肠型。在此,我们描述了一例完全内衬非角化鳞状上皮的降结肠重复畸形。因此,在我们的病例中结肠黏膜(内胚层起源)和鳞状上皮(外胚层来源)的关联是一个有趣的发现,各种理论均无法解释。此外,结合文献对肠道重复畸形的临床特征、诊断和治疗进行了讨论。

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