Ivo de Blaauw, Pernilla Stenström, Atsuyuki Yamataka, Yuichiro Miyake, Heiko Reutter, Paola Midrio, Richard Wood, Caterina Grano, Mikko Pakarinen
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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.</p>","PeriodicalId":18910,"journal":{"name":"Nature Reviews Disease Primers","volume":"10 1","pages":"88"},"PeriodicalIF":76.9000,"publicationDate":"2024-11-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Anorectal malformations.\",\"authors\":\"Ivo de Blaauw, Pernilla Stenström, Atsuyuki Yamataka, Yuichiro Miyake, Heiko Reutter, Paola Midrio, Richard Wood, Caterina Grano, Mikko Pakarinen\",\"doi\":\"10.1038/s41572-024-00574-2\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Anorectal malformations (ARM) are rare congenital anomalies with an overall prevalence of 3.32 per 10,000 pregnancies. 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引用次数: 0
摘要
肛门直肠畸形(ARM)是一种罕见的先天性畸形,总发病率为每万名孕妇中有 3.32 例。肛门直肠畸形描述了肛门和直肠的一系列畸形,从肛管轻微移位到肛门直肠、阴道和尿道完全融合,括约肌和盆底肌发育不良。妊娠第 6 至 9 周时,后肠的异常隔膜与异常泄殖腔膜构成了被定义为 ARM 的一系列畸形的发育基础。虽然潜在的特定综合征和偶尔出现的熟知病例提示了遗传病因,但大多数 ARM 并非综合征,其致病遗传机制和非遗传因素仍不清楚。ARM 是一种临床诊断,一般在出生后早期通过仔细检查会阴部而确诊。产前检查仍然很少见,现代技术的发展也没有为产前诊断带来多少帮助。ARM 可通过手术矫正。自 1982 年引入以来,后矢状位肛门成形术是最常见的 ARM 重建手术。后来的手术改良主要是通过限制手术创面来最大限度地减少手术造成的先天性损伤。这些手术包括腹腔镜手术和在开放手术中缩短切口并限制解剖范围。虽然 ARM 患者的治疗效果在过去几十年中有所改善,但仍迫切需要进一步提高功能效果和生活质量。受影响患者的社会心理经历的重要性日益得到认可。有必要继续开展研究,以改进产前检测,阐明遗传和表观遗传学改变,并完善最佳手术程序。
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.
期刊介绍:
Nature Reviews Disease Primers, a part of the Nature Reviews journal portfolio, features sections on epidemiology, mechanisms, diagnosis, management, and patient quality of life. The editorial team commissions top researchers — comprising basic scientists and clinical researchers — to write the Primers, which are designed for use by early career researchers, medical students and principal investigators. Each Primer concludes with an Outlook section, highlighting future research directions. Covered medical specialties include Cardiology, Dermatology, Ear, Nose and Throat, Emergency Medicine, Endocrinology, Gastroenterology, Genetic Conditions, Gynaecology and Obstetrics, Hepatology, Haematology, Infectious Diseases, Maxillofacial and Oral Medicine, Nephrology, Neurology, Nutrition, Oncology, Ophthalmology, Orthopaedics, Psychiatry, Respiratory Medicine, Rheumatology, Sleep Medicine, and Urology.