Potocki-Lupski syndrome: an inherited dup(17)(p11.2p11.2) with hypoplastic left heart

Am J Med Genet A. 2011 Feb;155A(2):367-71. doi: 10.1002/ajmg.a.33845.

Abstract

Low copy repeat (LCR) sequences in 17p11.2 predispose this region to genomic deletions and duplications. Duplication of 17p11.2, also known as Potocki-Lupski syndrome (PTLS), is a well-described microduplication syndrome featuring cognitive and language deficits, developmental delay, autistic behavior, structural cardiovascular anomalies, hypotonia, failure to thrive, apnea, and dysmorphism. We present a mother and her two children who share both dysmorphic features and the dup(17)(p11.2p11.2); the first child was born with hypoplastic left heart (HLH). The dup(17)(p11.2p11.2) was identified by GTG-banding analysis of peripheral blood specimens from all three individuals and confirmed by fluorescence in situ hybridization (FISH) and array comparative genomic hybridization (aCGH). Here we provide a thorough description of the phenotypes of the affected individuals, as well as describe physical features not reported previously for PTLS.

Publication types

  • Case Reports

MeSH terms

  • Abnormalities, Multiple
  • Child, Preschool
  • Chromosome Disorders
  • Chromosome Duplication
  • Comparative Genomic Hybridization
  • Female
  • Humans
  • Hypoplastic Left Heart Syndrome / pathology*
  • In Situ Hybridization, Fluorescence
  • Phenotype*
  • Smith-Magenis Syndrome / genetics
  • Smith-Magenis Syndrome / pathology

Supplementary concepts

  • Potocki-Lupski syndrome