| Literature DB >> 35040536 |
Catherine Gooch1, Jaclyn Paige Souder2, Matthew L Tedder3, Jennifer Kerkhof4, Jennifer A Lee3, Raymond J Louie3, Bekim Sadikovic4,5, Robin S Fletcher3, Nathaniel H Robin1.
Abstract
Pathogenic variants in KMT2D are typically associated with Kabuki syndrome (KS), a rare multisystem disorder. KS is characterized by facial dysmorphisms, intellectual disability, skeletal and dermatoglyphic differences, and poor growth. Seventy percent of individuals with clinically diagnosed KS have a confirmed pathogenic variant in KMT2D or less commonly KDM6A. The majority of mutations found in KMT2D are de novo nonsense or frameshift, with deletions and duplications rarely reported in the literature. Here, we present the case of near complete deletion of KMT2D in a college student with normal intelligence discovered via exome sequencing and EpiSign methylation testing. This case provides evidence that large deletions in KMT2D are compatible with normal intelligence and presents EpiSign as a method for discovering molecular causes of KS not identified by traditional molecular testing.Entities:
Keywords: KMT2D; Kabuki syndrome; deletion; molecular testing
Mesh:
Year: 2022 PMID: 35040536 PMCID: PMC8995339 DOI: 10.1002/ajmg.a.62652
Source DB: PubMed Journal: Am J Med Genet A ISSN: 1552-4825 Impact factor: 2.802