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[小儿神经病学进展]

[Progress in pediatric neurology].

作者信息

Landrieu P

机构信息

Service de Neuropédiatrie, Fédération de Pédiatrie, Fédération des Sciences Neurologiques et Psychiatriques, CHU Paris Sud-Bicêtre.

出版信息

Rev Neurol (Paris). 2000 Jul;156(6-7):595-605.

Abstract

A central part of Pediatric Neurology is currently dominated by the search for genetic factors involved in developmental disorders of the nervous system, including cases where the cytogenetic examination remains uncontributive. The prerequisite for a good definition of the malformative phenotypes leads to distinguish: 1 cerebral malformations that can be identified at the macroscopic scale, by imaging. 2 polymalformative syndromes including mental retardation where cerebral imaging is not contributive, thus the syndromatic definition is based on associated somatic anomalies. 3 Non-syndromatic mental retardation, where a genetic origin is clear only in the familial forms. Various methodological approaches have included genetic linkage studies, search for inframicroscopic chromosomal rearrangements in the critical region and investigation of candidate genes. A great number of syndromes have been connected with a great diversity of genetic mechanisms, whose many examples are presented: genopathies with regular or variable expression, unstable mutations, contiguous gene syndromes or other complex infracytogenetic rearrangements, chromosomal or genic mosaicisms, mutations submitted to parental imprinting. New methods of genomic screening will be necessary to progress in this field, given the great number of genes involved in cerebral development. As for the early developmental disorders of the PNS and muscle, their diagnosis becomes frequent during the intrauterine life, raising the problem of a better definition of the fetopathological phenotypes.

摘要

小儿神经病学的一个核心部分目前主要致力于寻找与神经系统发育障碍相关的遗传因素,包括细胞遗传学检查尚无结果的病例。准确界定畸形表型的前提是区分:1. 可通过影像学在宏观层面识别的脑畸形。2. 包括智力迟钝在内的多畸形综合征,此类综合征中脑影像学检查无诊断价值,因此综合征的定义基于相关的躯体异常。3. 非综合征性智力迟钝,仅在家族性病例中遗传起源明确。多种方法学途径包括遗传连锁研究、在关键区域寻找亚微观染色体重排以及对候选基因进行研究。大量综合征与多种遗传机制相关,文中列举了诸多实例:具有规律或可变表达的基因病、不稳定突变、相邻基因综合征或其他复杂的亚细胞遗传学重排、染色体或基因镶嵌现象、受亲本印记影响的突变。鉴于涉及脑发育的基因数量众多,在该领域取得进展需要新的基因组筛查方法。至于周围神经系统和肌肉的早期发育障碍,其诊断在子宫内生活期间变得越来越常见,这就引发了如何更好地界定胎儿病理表型的问题。

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