Phenotypic heterogeneity and mosaicism in Xia-Gibbs syndrome: Five Danish patients with novel variants in AHDC1

Research output: Contribution to journalJournal articleResearchpeer-review

  • Soren L. Faergeman
  • Anders B. Bojesen
  • Maria Rasmussen
  • Naja Becher
  • Lotte Andreasen
  • Brian N. Andersen
  • Emilie Erbs
  • Dorte L. Lildballe
  • Nielsen, Jens Erik Klint
  • Monica Zilmer
  • Trine Bjørg Hammer
  • Mikkel Andersen
  • Charlotte Brasch-Andersen
  • Christina R. Fagerberg
  • Niels O. Illum
  • Mette B. Thorup
  • Pernille A. Gregersen

Xia-Gibbs syndrome (XGS) is a neurodevelopmental disorder characterized by intellectual disability, developmental delay, seizures, hypotonia, obstructive sleep apnoea and mild facial dysmorphism. Heterozygosity for loss-of-function variants in AHDC1, encoding the AT-hook DNA binding motif containing protein 1, were discovered in 2014 as the likely genetic cause of Xia-Gibbs syndrome. We present five patients with Xia-Gibbs syndrome caused by previously unreported variants in AHDC1. Two of the patients share a frameshift variant: c.2849del (p.(Pro950Argfs*192)) in AHDC1. Despite sharing this variant, the two patients show remarkable phenotypic differences underscoring the clinical heterogeneity of Xia-Gibbs syndrome. In addition, we present a case of Xia-Gibbs syndrome caused by mosaicism for an AHDC1 variant.

Original languageEnglish
Article number104280
JournalEuropean Journal of Medical Genetics
Volume64
Issue number9
ISSN1769-7212
DOIs
Publication statusPublished - Sep 2021

Bibliographical note

Publisher Copyright:
© 2021

    Research areas

  • AHDC1, Dysmorphism, Reverse phenotyping, Whole exome sequencing, Xia-gibbs syndrome

ID: 305534195