NSDHL-containing duplication at Xq28 in a male patient with autism spectrum disorder: a case report

BMC Med Genet. 2018 Oct 30;19(1):192. doi: 10.1186/s12881-018-0705-7.

Abstract

Background: Autism spectrum disorder (ASD) is a neurodevelopmental disorder in which genetics plays a key aetiological role. The gene encoding NAD(P)H steroid dehydrogenase-like protein (NSDHL) is expressed in developing cortical neurons and glia, and its mutation may result in intellectual disability or congenital hemidysplasia.

Case presentation: An 8-year-old boy presented with a 260-kb NSDHL-containing duplication at Xq28 (151,868,909 - 152,129,300) inherited from his mother. His clinical features included defects in social communication and interaction, restricted interests, attention deficit, impulsive behaviour, minor facial anomalies and serum free fatty acid abnormality.

Conclusion: This is the first report of an ASD patient with a related NSDHL-containing duplication at Xq28. Further studies and case reports are required for genetic research to demonstrate that duplication as well as mutation can cause neurodevelopmental diseases.

Keywords: Autism; CNV; NSDHL; Xq28 duplication.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • 3-Hydroxysteroid Dehydrogenases / genetics*
  • Adult
  • Autism Spectrum Disorder / blood
  • Autism Spectrum Disorder / diagnosis
  • Autism Spectrum Disorder / genetics*
  • Autism Spectrum Disorder / physiopathology
  • Child
  • Chromosome Duplication*
  • Chromosomes, Human, Pair 10 / chemistry*
  • Fatty Acids, Nonesterified / blood
  • Female
  • Gene Dosage
  • Gene Expression
  • Humans
  • Male
  • Maternal Inheritance*

Substances

  • Fatty Acids, Nonesterified
  • 3-Hydroxysteroid Dehydrogenases
  • Nsdhl protein, human