Literature DB >> 24727054

Mosaicism for c.431_454dup in ARX causes a mild Partington syndrome phenotype.

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.
Copyright © 2014 Elsevier Masson SAS. All rights reserved.

Entities:  

Keywords:  ARX; DNA sequencing; Duplication; Mosaicism; PCR fragment length analysis; Partington syndrome

Mesh:

Substances:

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


  2 in total

1.  A novel mutation in the OAR domain of the ARX gene.

Authors:  Alejandra Tapie; Natalia Pi-Denis; Jorge Souto; Alejandra Vomero; Gabriel Peluffo; María Boidi; Martín Ciganda; Nicolás Curbelo; Victor Raggio; Leda Roche; Lucía Pastro
Journal:  Clin Case Rep       Date:  2017-01-23

2.  Disease-associated mosaic variation in clinical exome sequencing: a two-year pediatric tertiary care experience.

Authors:  Cecelia R Miller; Kristy Lee; Ruthann B Pfau; Shalini C Reshmi; Donald J Corsmeier; Sayaka Hashimoto; Ashita Dave-Wala; Vijayakumar Jayaraman; Daniel Koboldt; Theodora Matthews; Danielle Mouhlas; Maggie Stein; Aimee McKinney; Tom Grossman; Benjamin J Kelly; Peter White; Vincent Magrini; Richard K Wilson; Elaine R Mardis; Catherine E Cottrell
Journal:  Cold Spring Harb Mol Case Stud       Date:  2020-06-12
  2 in total

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