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整合分子和结构研究发现:Wnt 可能是 Cornelia de Lange 综合征导致认知障碍的一个作用因子。

Integrating molecular and structural findings: Wnt as a possible actor in shaping cognitive impairment in Cornelia de Lange syndrome.

机构信息

Department of Health Sciences, San Paolo Hospital Medical School University of Milan, Via A. di Rudinì, 8, 20142, Milan, Italy.

UOC Pediatria, ASST Lariana, Como, Italy.

出版信息

Orphanet J Rare Dis. 2017 Nov 21;12(1):174. doi: 10.1186/s13023-017-0723-0.

Abstract

Cornelia de Lange Syndrome (CdLS) is a choesinopathy: a severe genetic disorder caused by mutations in the cohesin complex genes. The phenotype is characterized by typical facial dysmorphism, growth impairment and multiorgan abnormalities including brain alterations. Wnt pathway is known to play a fundamental role in central nervous system development and it has been shown that Wnt pathway is disrupted in CdLS animal models and patients cells. In this review we investigate the possible link between Wnt pathway disruption and brain abnormalities in Cornelia de Lange Syndrome as such molecular impairment could lead to an abnormal embryonic development resulting in brain abnormalities (i.e. microcephaly, cerebellar hypoplasia, abnormal cortical development) in patients with Cornelia de Lange Syndrome.

摘要

康氏综合征(CdLS)是一种黏连蛋白病:一种由黏连蛋白复合物基因突变引起的严重遗传性疾病。其表型特征为典型的面部畸形、生长障碍和多器官异常,包括大脑改变。Wnt 通路已知在中枢神经系统发育中起重要作用,并且已经表明 Wnt 通路在 CdLS 动物模型和患者细胞中被破坏。在本综述中,我们研究了 Wnt 通路破坏与康氏综合征大脑异常之间的可能联系,因为这种分子损伤可能导致异常的胚胎发育,从而导致康氏综合征患者的大脑异常(如小头畸形、小脑发育不全、皮质发育异常)。

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