A novel de novo CASZ1 heterozygous frameshift variant causes dilated cardiomyopathy and left ventricular noncompaction cardiomyopathy

Mol Genet Genomic Med. 2019 Aug;7(8):e828. doi: 10.1002/mgg3.828. Epub 2019 Jul 3.

Abstract

Background: Dilated cardiomyopathy (DCM) is the most common cardiomyopathy with a common presentation of heart failure. It has been reported that CASZ1 loss-of-function mutation contributes to familial DCM and congenital ventricular septal defect (VSD). To date, only two pathogenic variants in CASZ1 have been previously reported worldwide.

Methods: To identify the causative variant in an 11-month-old Chinese boy with DCM and left ventricular noncompaction cardiomyopathy (LVNC), trio-whole-exome sequencing was performed followed by mutational analysis and Sanger sequencing.

Results: An unreported de novo heterozygous frameshift variant (c.2443_2459delGTGGGCACCCCCAGCCT, p.Val815Profs*14) in CASZ1 was idenitified in the proband. The frameshift mutation in CASZ1 not only led to DCM but also presented an LVNC phenotype.

Conclusion: We have identified a novel CASZ1 variant in a patient with combined DCM and LVNC for the first time, thus broadening the phenotypic spectrum of CASZ1 variants. Furthermore, this study emphasized the usefulness of whole-exome sequencing for genetic diagnosis of cardiomyopathy.

Keywords: CASZ1 variant; DCM; LVNC; whole-exome sequencing.

Publication types

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

MeSH terms

  • Asian People / genetics
  • Cardiomyopathy, Dilated / complications
  • Cardiomyopathy, Dilated / diagnosis
  • Cardiomyopathy, Dilated / genetics*
  • DNA Mutational Analysis
  • DNA-Binding Proteins / genetics*
  • Echocardiography
  • Exome Sequencing
  • Fatal Outcome
  • Frameshift Mutation
  • Heart / diagnostic imaging
  • Heterozygote
  • Humans
  • Infant
  • Isolated Noncompaction of the Ventricular Myocardium / complications
  • Isolated Noncompaction of the Ventricular Myocardium / diagnosis
  • Isolated Noncompaction of the Ventricular Myocardium / genetics*
  • Male
  • Transcription Factors / genetics*
  • Ventricular Fibrillation / etiology*

Substances

  • CASZ1 protein, human
  • DNA-Binding Proteins
  • Transcription Factors

Supplementary concepts

  • Cardiomyopathy, Dilated, with Left Ventricular Noncompaction