Literature DB >> 28742278

Agenesis of the corpus callosum, developmental delay, autism spectrum disorder, facial dysmorphism, and posterior polymorphous corneal dystrophy associated with ZEB1 gene deletion.

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.
© 2017 Wiley Periodicals, Inc.

Entities:  

Keywords:  ZEB1; agenesis of corpus callosum (ACC); array-CGH; autism spectrum disorder; chromosome 10p11.22; corneal dystrophy; deletion

Mesh:

Substances:

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


  3 in total

1.  Non-Penetrance for Ocular Phenotype in Two Individuals Carrying Heterozygous Loss-of-Function ZEB1 Alleles.

Authors:  Lubica Dudakova; Viktor Stranecky; Lenka Piherova; Tomas Palecek; Nikolas Pontikos; Stanislav Kmoch; Pavlina Skalicka; Manuela Vaneckova; Alice E Davidson; Petra Liskova
Journal:  Genes (Basel)       Date:  2021-04-30       Impact factor: 4.096

2.  Integrated Analysis of Brain Transcriptome Reveals Convergent Molecular Pathways in Autism Spectrum Disorder.

Authors:  Xiaodan Li; Yuncong Zhang; Luxi Wang; Yunqing Lin; Zhaomin Gao; Xiaolei Zhan; Yan Huang; Caihong Sun; Dong Wang; Shuang Liang; Lijie Wu
Journal:  Front Psychiatry       Date:  2019-10-08       Impact factor: 4.157

Review 3.  Update on the genetics of corneal endothelial dystrophies.

Authors:  Chitra Kannabiran; Sunita Chaurasia; Muralidhar Ramappa; Venkateswara Vinod Mootha
Journal:  Indian J Ophthalmol       Date:  2022-07       Impact factor: 2.969

  3 in total

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