| Literature DB >> 24727054 |
Karen Grønskov1, Birgitte Diness2, Michelle Stahlhut3, Monica Zilmer4, Zeynep Tümer5, Anne-Marie Bisgaard3, Karen Brøndum-Nielsen5.
Abstract
A common in frame duplication in ARX (c.431_454dup24) was found in a five year-old boy who presented with mild Partington syndrome. The duplication was detected by PCR amplification followed by fragment length analysis and was located in exon 2 spanning the two polyalanine tracts commonly seen to expand. Detection of the duplication by DNA sequencing was difficult due to preferential sequencing of the normal allele, demonstrating the superiority of fragment length analysis in mosaic cases. The clinical symptoms were mild to moderate developmental delay with only the hand dystonia to suggest Partington syndrome. This patient is the first male reported to be mosaic for the duplication, and his clinical features are subtle. This study shows that in males with a phenotype of mild Partington syndrome and in heterozygous females fragment length analysis should be preferred over DNA sequencing.Entities:
Keywords: ARX; DNA sequencing; Duplication; Mosaicism; PCR fragment length analysis; Partington syndrome
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Year: 2014 PMID: 24727054 DOI: 10.1016/j.ejmg.2014.03.009
Source DB: PubMed Journal: Eur J Med Genet ISSN: 1769-7212 Impact factor: 2.708