| Literature DB >> 23713105 |
Rebecca E Rosenberg1, Maureen Egan, Shaun Rodgers, David Harter, Rachel D Burnside, Sarah Milla, John Pappas.
Abstract
A 7-year-old white girl presented with left hemiparesis and ischemic stroke secondary to moyamoya syndrome, a progressive cerebrovascular occlusive disorder of uncertain but likely multifactorial etiology. Past medical history revealed hearing loss and developmental delay/intellectual disability. Routine karyotype demonstrated extra chromosomal material on 6p. Single nucleotide polymorphism microarray revealed a previously unreported complex de novo genetic rearrangement involving subtelomeric segments on chromosomes 6p and 12q. The duplicated/deleted regions included several known OMIM-annotated genes. This novel phenotype and genotype provides information about a possible association of genomic copy number variation and moyamoya syndrome. Dosage-sensitive genes in the deleted and duplicated segments may be involved in aberrant vascular proliferation. Our case also emphasizes the importance of comprehensive evaluation of both developmental delay and congenital anomalies such as moyamoya.Entities:
Keywords: 12q deletion; 6p trisomy; moyamoya
Mesh:
Year: 2013 PMID: 23713105 DOI: 10.1542/peds.2012-0749
Source DB: PubMed Journal: Pediatrics ISSN: 0031-4005 Impact factor: 7.124