| Literature DB >> 19034313 |
Sandesh Chakravarthy Sreenath Nagamani1, Ayelet Erez, Christine Eng, Zhishuo Ou, Craig Chinault, Laura Workman, James Coldwell, Pawel Stankiewicz, Ankita Patel, James R Lupski, Sau Wai Cheung.
Abstract
Interstitial deletions of 6q are rare. We report a detailed clinical and molecular characterization of four patients with interstitial deletion involving 6q25. All of our patients presented with microcephaly, developmental delay, dysmorphic features and hearing loss, whereas two of them had agenesis of the corpus callosum. We determined the size, extent and genomic content of the deletions using high-density array-comparative genomic hybridization (a-CGH), and found that a common segment spanning 3.52 Mb within the 6q25.2-q25.3 region was deleted in all four cases. We hypothesize that a subset of genes in the commonly deleted region are dosage sensitive and that haploinsufficieny of these genes impairs normal development of the brain and hearing.Entities:
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Year: 2008 PMID: 19034313 PMCID: PMC2986272 DOI: 10.1038/ejhg.2008.220
Source DB: PubMed Journal: Eur J Hum Genet ISSN: 1018-4813 Impact factor: 4.246