| Literature DB >> 28742278 |
Ayeshah Chaudhry1,2, Brian H Chung3, Dimitri J Stavropoulos4, Marcela P Araya5, Asim Ali5, Elise Heon5, David Chitayat6,7.
Abstract
We report on a girl diagnosed prenatally with agenesis of the corpus callosum (ACC) on fetal ultrasound and MRI. On postnatal follow-up she was noted to have developmental delay, facial dysmorphism, autism spectrum disorder, and posterior polymorphous corneal dystrophy (PPD). Array-comparative genomic hybridization analysis (Array-CGH) showed a 2.05 Mb de novo interstitial deletion at 10p11.23p11.22. The deleted region overlaps 1 OMIM Morbid Map gene, ZEB1 (the zinc finger E-box binding homeobox transcription factor 1), previously associated with posterior polymorphous corneal dystrophy type 3 (PPCD3). To our best knowledge this is the first reported case with a deletion of the ZEB1 gene in an individual with ACC and PPD, showing that the haploinsufficiency of the ZEB1 is likely the cause of our patient's phenotype.Entities:
Keywords: ZEB1; agenesis of corpus callosum (ACC); array-CGH; autism spectrum disorder; chromosome 10p11.22; corneal dystrophy; deletion
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Year: 2017 PMID: 28742278 DOI: 10.1002/ajmg.a.38321
Source DB: PubMed Journal: Am J Med Genet A ISSN: 1552-4825 Impact factor: 2.802