Novel Mutations Segregating with Complete Androgen Insensitivity Syndrome and their Molecular Characteristics

Int J Mol Sci. 2019 Oct 30;20(21):5418. doi: 10.3390/ijms20215418.

Abstract

We analyzed three cases of Complete Androgen Insensitivity Syndrome (CAIS) and report three hitherto undisclosed causes of the disease. RNA-Seq, Real-timePCR, Western immunoblotting, and immunohistochemistry were performed with the aim of characterizing the disease-causing variants. In case No.1, we have identified a novel androgen receptor (AR) mutation (c.840delT) within the first exon in the N-terminal transactivation domain. This thymine deletion resulted in a frameshift and thus introduced a premature stop codon at amino acid 282. In case No.2, we observed a nonsynonymous mutation in the ligand-binding domain (c.2491C>T). Case No.3 did not reveal AR mutation; however, we have found a heterozygous mutation in CYP11A1 gene, which has a role in steroid hormone biosynthesis. Comparative RNA-Seq analysis of CAIS and control revealed 4293 significantly deregulated genes. In patients with CAIS, we observed a significant increase in the expression levels of PLCXD3, TM4SF18, CFI, GPX8, and SFRP4, and a significant decrease in the expression of SPATA16, TSACC, TCP10L, and DPY19L2 genes (more than 10-fold, p < 0.05). Our findings will be helpful in molecular diagnostics of patients with CAIS, as well as the identified genes could be also potential biomarkers for the germ cells differentiation process.

Keywords: Morris syndrome; RNA-seq; androgen insensitivity syndrome; biomarkers; infertility; rare disease; spermatogenesis.

Publication types

  • Case Reports

MeSH terms

  • Adolescent
  • Adult
  • Androgen-Insensitivity Syndrome / genetics*
  • Androgen-Insensitivity Syndrome / metabolism
  • Case-Control Studies
  • Cholesterol Side-Chain Cleavage Enzyme / genetics*
  • Cholesterol Side-Chain Cleavage Enzyme / metabolism
  • Exons
  • Female
  • Frameshift Mutation
  • Gene Expression Profiling
  • Gene Expression Regulation
  • Gene Regulatory Networks
  • Humans
  • Male
  • Middle Aged
  • Mutation*
  • Polymorphism, Single Nucleotide
  • Protein Domains
  • Receptors, Androgen / chemistry
  • Receptors, Androgen / genetics*
  • Receptors, Androgen / metabolism
  • Sequence Analysis, DNA / methods*
  • Young Adult

Substances

  • AR protein, human
  • Receptors, Androgen
  • Cholesterol Side-Chain Cleavage Enzyme