新生儿先天性心脏病的单基因综合征(新生儿学家的诊断线索):系统文献综述的关键分析。

IF 0.4 Q4 PEDIATRICS Journal of pediatric genetics Pub Date : 2021-09-01 Epub Date: 2021-07-10 DOI:10.1055/s-0041-1731036
Raffaele Falsaperla, Valentina Giacchi, Maria Giovanna Aguglia, Janette Mailo, Maria Grazia Longo, Federica Natacci, Martino Ruggieri
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引用次数: 1

摘要

先天性心脏病(CHD)是最常见的主要先天性异常,在30%的患者中与遗传综合征(染色体异常、基因组疾病或单基因疾病)相关。本系统综述的目的是评估是否,在新生儿设置,临床线索,定向诊断路径可以确定。为此,我们修订了冠心病新生儿中最常见的畸形特征,将与单基因综合征(MSG)相关的畸形特征与基因组疾病新生儿中报道的畸形特征进行了比较。根据PRISMA声明,我们使用PubMed, Medline, Google Scholar, Scopus数据库,检索与冠心病及其综合征相关的关键词。我们发现,在半数以上的msg中检测到的广泛的畸形(眼区、耳区、嘴区和/或腭区和指骨异常)被发现与冠心病有关,但这些异常也被描述为同样普遍的基因组重排综合征。这些发现证实,新生儿的病因诊断是具有挑战性的,只有及时和专家识别暗示遗传条件的特征,才能改善选择适当的、具有成本效益的诊断测试。然而,在一般实践中,识别遗传综合征存在的线索是至关重要的,新生儿科医生通常有独特的机会第一个发现新生儿的异常。
本文章由计算机程序翻译,如有差异,请以英文原文为准。

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Monogenic Syndromes with Congenital Heart Diseases in Newborns (Diagnostic Clues for Neonatologists): A Critical Analysis with Systematic Literature Review.

Congenital heart disease (CHD), the most common major congenital anomaly, is associated with a genetic syndrome (chromosomal anomalies, genomic disorders, or monogenic disease) in 30% of patients. The aim of this systematic review was to evaluate if, in the neonatal setting, clinical clues that orient the diagnostic path can be identified. For this purpose, we revised the most frequent dysmorphic features described in newborns with CHD, comparing those associated with monogenic syndromes (MSG) with the ones reported in newborns with genomic disorders. For this systematic review according to PRISMA statement, we used PubMed, Medline, Google Scholar, Scopus database, and search terms related to CHD and syndrome. We found a wide range of dysmorphisms (ocular region, ears, mouth, and/or palate and phalangeal anomalies) detected in more than half of MSGs were found to be associated with CHDs, but those anomalies are also described in genomic rearrangements syndromes with equal prevalence. These findings confirmed that etiological diagnosis in newborns is challenging, and only the prompt and expert recognition of features suggestive of genetic conditions can improve the selection of appropriate, cost-effective diagnostic tests. However, in general practice, it is crucial to recognize clues that can suggest the presence of a genetic syndrome, and neonatologists often have the unique opportunity to be the first to identify abnormalities in the neonate.

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期刊介绍: The Journal of Pediatric Genetics is an English multidisciplinary peer-reviewed international journal publishing articles on all aspects of genetics in childhood and of the genetics of experimental models. These topics include clinical genetics, molecular genetics, biochemical genetics, medical genetics, dysmorphology, teratology, genetic counselling, genetic engineering, formal genetics, neuropsychiatric genetics, behavioral genetics, community genetics, cytogenetics, hereditary or syndromic cancer genetics, genetic mapping, reproductive genetics, fetal pathology and prenatal diagnosis, multiple congenital anomaly syndromes, and molecular embryology of birth defects. Journal of Pediatric Genetics provides an in-depth update on new subjects and current comprehensive coverage of the latest techniques used in the diagnosis of childhood genetics. Journal of Pediatric Genetics encourages submissions from all authors throughout the world. The following articles will be considered for publication: editorials, original and review articles, short report, rapid communications, case reports, letters to the editor, and book reviews. The aim of the journal is to share and disseminate knowledge between all disciplines in the field of pediatric genetics. This journal is a publication of the World Pediatric Society: http://www.worldpediatricsociety.org/ The Journal of Pediatric Genetics is available in print and online. Articles published ahead of print are available via the eFirst service on the Thieme E-Journals platform.
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