On the significance of craniosynostosis in a case of Kabuki syndrome with a concomitant KMT2D mutation and 3.2 Mbp de novo 10q22.3q23.1 deletion

Am J Med Genet A. 2017 Aug;173(8):2219-2225. doi: 10.1002/ajmg.a.38296. Epub 2017 Jun 7.

Abstract

Craniosynostosis has rarely been described in patients with Kabuki syndrome. We report here a boy with facial asymmetry due to combined premature synostosis of the right coronal and sagittal sutures as well as several symptoms reminiscent of Kabuki syndrome (KS). Our case supports previous observations and suggests that craniosynostosis is a part of the KS phenotype. The uniqueness of our case is the sporadic co-occurrence of two genetic disorders, that is, a de novo frameshift variant in the KMT2D gene and a de novo 3.2 Mbp 10q22.3q23.1 deletion. Our findings emphasize the importance of the initial clinical assessment of children with craniosynostosis and that genomic and monogenic disorders, such as Kabuki syndrome, should be considered among the differential diagnoses of syndromic forms of craniosynostosis.

Keywords: 10q deletion; KMT2D; Kabuki syndrome; craniosynostosis.

Publication types

  • Case Reports

MeSH terms

  • Abnormalities, Multiple / genetics*
  • Abnormalities, Multiple / physiopathology
  • Child
  • Chromosome Deletion
  • Chromosomes, Human, Pair 10 / genetics
  • Craniosynostoses / complications
  • Craniosynostoses / genetics*
  • Craniosynostoses / physiopathology
  • DNA-Binding Proteins / genetics*
  • Face / abnormalities*
  • Face / physiopathology
  • Hematologic Diseases / complications
  • Hematologic Diseases / genetics*
  • Hematologic Diseases / physiopathology
  • Humans
  • Male
  • Mutation
  • Neoplasm Proteins / genetics*
  • Phenotype
  • Sequence Deletion
  • Vestibular Diseases / complications
  • Vestibular Diseases / genetics*
  • Vestibular Diseases / physiopathology

Substances

  • DNA-Binding Proteins
  • KMT2D protein, human
  • Neoplasm Proteins

Supplementary concepts

  • Chromosome 10, monosomy 10q
  • Kabuki syndrome