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临床问答——库拉里诺综合征背景下的罕见肛管重复畸形病例

Clinical Quiz-A Rare Case of Anal Canal Duplication in the Context of Currarino Syndrome.

作者信息

Connor Sean Jared, Brisighelli Giulia, Patel Nirav, Levitt Marc A

机构信息

Department of Paediatric Surgery, Chris Hani Baragwanath Hospital, Johannesburg, Gauteng, South Africa.

University of the Witwatersrand School of Clinical Medicine, Johannesburg, Gauteng, South Africa.

出版信息

European J Pediatr Surg Rep. 2021 Nov 10;9(1):e68-e71. doi: 10.1055/s-0041-1735595. eCollection 2021 Jan.

DOI:10.1055/s-0041-1735595
PMID:34777941
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC8580735/
Abstract

Currarino syndrome (CS) is a rare condition that presents with any combination of a sacral defect, a presacral mass, and an anorectal malformation. This collection, referred to as Currarino's triad, may not necessarily present as all three abnormalities in the diagnosis of the syndrome. Anal canal duplication (ACD) is an even rarer occurrence. We present a case that lies on the CS spectrum with an associated ACD and discuss a complex surgical challenge that necessitated a customized management plan, devised through a multidisciplinary approach.

摘要

库拉里诺综合征(CS)是一种罕见病症,表现为骶骨缺损、骶前肿块和肛门直肠畸形的任意组合。这种组合,即所谓的库拉里诺三联征,在该综合征的诊断中不一定会同时出现所有三种异常情况。肛管重复畸形(ACD)则更为罕见。我们报告一例属于CS谱系且伴有ACD的病例,并讨论了一个复杂的手术挑战,这需要通过多学科方法制定定制化的管理方案。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/62dd/8580735/a9e4b712e086/10-1055-s-0041-1735595-i210591-1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/62dd/8580735/a9e4b712e086/10-1055-s-0041-1735595-i210591-1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/62dd/8580735/a9e4b712e086/10-1055-s-0041-1735595-i210591-1.jpg

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本文引用的文献

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Neurosurgical management of Currarino syndrome: A case series and review of literature.库拉里诺综合征的神经外科治疗:病例系列及文献综述
Surg Neurol Int. 2019 Apr 24;10:70. doi: 10.25259/SNI-26-2019. eCollection 2019.
2
Anal duplication: is surgery indicated? A report of three cases and review of the literature.
Pediatr Surg Int. 2019 Sep;35(9):971-978. doi: 10.1007/s00383-019-04509-x. Epub 2019 Jun 29.
3
Differentiating presacral masses in anorectal malformations and isolated sacrococcygeal teratomas.鉴别肛门直肠畸形和孤立性骶尾部畸胎瘤中的骶前肿物。
Pediatr Surg Int. 2019 Sep;35(9):979-983. doi: 10.1007/s00383-019-04510-4. Epub 2019 Jun 29.
4
Anal canal duplication and triplication: a rare entity with different presentations.肛管重复畸形和三联畸形:一种具有不同表现的罕见病症。
Pediatr Surg Int. 2017 May;33(5):609-617. doi: 10.1007/s00383-017-4074-7. Epub 2017 Mar 2.
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Currarino Syndrome in a Fetus, Infant, Child, and Adolescent: Spectrum of Clinical Presentations and Imaging Findings.胎儿、婴儿、儿童及青少年的库拉里诺综合征:临床表现及影像学表现谱
Can Assoc Radiol J. 2017 Feb;68(1):90-95. doi: 10.1016/j.carj.2016.05.007. Epub 2016 Nov 22.
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Anal canal duplication: experience at a single institution and literature review.肛管重复畸形:单机构经验及文献综述
Pediatr Surg Int. 2010 Oct;26(10):985-8. doi: 10.1007/s00383-010-2653-y.
7
Anal canal duplication: case reviews and summary of the world literature.肛管重复畸形:病例回顾及世界文献综述
Pediatr Surg Int. 2009 Oct;25(10):911-6. doi: 10.1007/s00383-009-2474-z.
8
Anal canal duplication: a retrospective analysis of 12 cases from two European pediatric surgical departments.肛管重复畸形:来自两个欧洲小儿外科科室的12例病例回顾性分析
Pediatr Surg Int. 2006 Dec;22(12):967-73. doi: 10.1007/s00383-006-1801-x.
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Pediatr Surg Int. 2006 Jun;22(6):560-1. doi: 10.1007/s00383-006-1654-3. Epub 2006 Mar 15.
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Pediatr Surg Int. 2002 Mar;18(2-3):195-7. doi: 10.1007/s003830100691.