Integrating molecular and structural findings: Wnt as a possible actor in shaping cognitive impairment in Cornelia de Lange syndrome.
Laura AvaglianoPaolo GrazioliMilena MarianiGaetano P BulfamanteAngelo SelicorniValentina MassaPublished in: Orphanet journal of rare diseases (2017)
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.
Keyphrases
- cell proliferation
- resting state
- stem cells
- case report
- white matter
- cognitive impairment
- end stage renal disease
- functional connectivity
- genome wide
- chronic kidney disease
- induced apoptosis
- newly diagnosed
- early onset
- gene expression
- prognostic factors
- cell cycle arrest
- signaling pathway
- autism spectrum disorder
- blood brain barrier
- drug induced
- cell death
- soft tissue