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肛门直肠畸形。

Anorectal malformations.

机构信息

Department of Surgery, Division of Paediatric Surgery, Radboudumc-Amalia Children's Hospital, Nijmegen, the Netherlands.

Department of Paediatric Surgery, Institution of Clinical Sciences, Lund university, Skane University Hospital, Lund, Sweden.

出版信息

Nat Rev Dis Primers. 2024 Nov 21;10(1):88. doi: 10.1038/s41572-024-00574-2.

DOI:10.1038/s41572-024-00574-2
PMID:39572572
Abstract

Anorectal malformations (ARM) are rare congenital anomalies with an overall prevalence of 3.32 per 10,000 pregnancies. ARM describe a spectrum of anomalies of the anus and rectum ranging from a minimally displaced anal canal to a complete fusion of the anorectum, vagina and urethra with hypoplastic sphincter and pelvic floor muscle. Aberrant septation of the hindgut with anomalous cloacal membrane during weeks 6 to 9 of gestation form the developmental basis for a spectrum of anomalies defined as ARM. Although underlying specific syndromes and occasional familiar occurrence suggest genetic aetiology, most ARM are non-syndromic and their causal genetic mechanisms and non-genetic insults remain unclear. ARM is a clinical diagnosis, generally made early after birth via careful inspection of the perineum. Prenatal detection remains rare, and modern technical developments have added little to prenatal diagnostics. ARM is corrected surgically. Since its introduction in 1982, posterior sagittal anorectoplasty is the most common surgery for ARM reconstruction. Subsequent surgical adaptations focus on minimizing iatrogenic operative injury by limiting surgical invasiveness. They include laparoscopic procedures and shortening of incisions with confined dissection in open surgery. Although outcomes in patients with ARM have evolved throughout the past decades, there is urgent need for further improvements both in functional outcomes and quality of life. The importance of psychosocial experiences of affected patients is increasingly recognized. Continued research is necessary to improve prenatal detection, to elucidate genetic and epigenetic alterations and to refine optimal surgical procedures.

摘要

肛门直肠畸形(ARM)是一种罕见的先天性畸形,其总体患病率为每 10000 例妊娠中 3.32 例。ARM 描述了肛门和直肠的一系列异常,从轻微移位的肛管到肛门、直肠、阴道和尿道完全融合,伴有发育不良的括约肌和盆底肌肉。在妊娠第 6 至 9 周期间,后肠异常分隔和异常的膜性肛膜形成了定义为 ARM 的一系列异常的发育基础。尽管特定综合征的潜在异常和偶尔的家族性发生提示遗传病因,但大多数 ARM 是非综合征性的,其因果遗传机制和非遗传损伤仍不清楚。ARM 是一种临床诊断,通常在出生后早期通过仔细检查会阴部来做出。产前检测仍然很少见,现代技术发展对产前诊断几乎没有增加。ARM 通过手术进行矫正。自 1982 年引入以来,后路矢状位肛门直肠成形术是 ARM 重建最常见的手术。随后的手术适应主要通过限制手术侵袭性来最大限度地减少医源性手术损伤。它们包括腹腔镜手术和开放性手术中切口缩短和限制解剖。尽管过去几十年 ARM 患者的治疗效果有所发展,但在功能结果和生活质量方面仍迫切需要进一步改善。受影响患者的心理社会体验的重要性日益得到认识。需要继续研究以提高产前检测水平,阐明遗传和表观遗传改变,并完善最佳手术程序。

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Gastroenterol Clin North Am. 2024 Sep;53(3):461-472. doi: 10.1016/j.gtc.2023.12.006. Epub 2024 Jan 23.
2
High-grade Vesicoureteral Reflux in Patients With Anorectal Malformation From the ARM-Net Registry: Is Our Screening Sufficient?来自肛门直肠畸形网络注册研究(ARM-Net Registry)的肛门直肠畸形患者的重度膀胱输尿管反流:我们的筛查是否足够?
J Pediatr Surg. 2024 Aug;59(8):1470-1476. doi: 10.1016/j.jpedsurg.2024.01.008. Epub 2024 Jan 19.
3
Treatment of Anorectal Malformations in German Hospitals: Analysis of National Hospital Discharge Data from 2016 to 2021.
德国医院的肛门直肠畸形治疗:2016 年至 2021 年全国住院数据分析。
Eur J Pediatr Surg. 2024 Dec;34(6):501-511. doi: 10.1055/a-2260-5124. Epub 2024 Feb 2.
4
Genetically proxied low-density lipoprotein cholesterol lowering via PCSK9-inhibitor drug targets and risk of congenital malformations.通过 PCSK9 抑制剂药物靶点降低遗传上接近的低密度脂蛋白胆固醇与先天性畸形风险。
Eur J Prev Cardiol. 2024 Jun 3;31(8):955-965. doi: 10.1093/eurjpc/zwad402.
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Functional Outcomes of Patients Who Underwent Anorectal Malformation Repair Using MRI Guidance.采用 MRI 引导的肛门直肠畸形修复术患者的功能结果。
J Pediatr Surg. 2024 Jun;59(6):1044-1049. doi: 10.1016/j.jpedsurg.2023.12.023. Epub 2024 Jan 1.
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Adult urological outcomes for patients with anorectal malformation.成人肛肠畸形患者的泌尿系统结局。
J Pediatr Urol. 2024 Apr;20(2):193-199. doi: 10.1016/j.jpurol.2023.12.015. Epub 2023 Dec 22.
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