[Phenotypic and genetic analysis of a pedigree with 4p16 microduplication and 8p23 microdeletion]

Zhonghua Yi Xue Yi Chuan Xue Za Zhi. 2019 Oct 10;36(10):989-992. doi: 10.3760/cma.j.issn.1003-9406.2019.10.009.
[Article in Chinese]

Abstract

Objective: To explore the nature and origin of chromosomal copy number variants (CNVs) in a pedigree affected with mental retardation.

Methods: Genomic CNVs of the proband were analyzed by next generation sequencing (NGS). Chromosomal karyotypes of the proband and his relatives were analyzed with high-resolution karyotyping and fluorescence in situ hybridization (FISH).

Results: Clinical phenotypes of the proband and other patients from the pedigree included mental retardation and mild dysmorphism. The results of NGS revealed that the proband carried a 16.24 Mb microduplication at 4p16.3-15.32 and a 2.2 Mb microdeletion at 8p23.3-23.2. Other patients of the pedigree harbored the same variants, while those without the phenotypes did not harbor the variants. The results of high-resolution karyotyping and FISH revealed that the mother of the proband carried a reciprocal translocation between 4p and 8p, and her karyotype was 46,XX,t(4;8)(p16;p23). No karyotypic abnormality was detected in his father.

Conclusion: The abnormal phenotypes of this pedigree may be attributed to 4p microduplication in conjunct with 8p microdeletion derived from a maternal balanced translocation between 4p and 8p.

MeSH terms

  • Chromosome Aberrations*
  • Chromosome Duplication*
  • Chromosomes, Human, Pair 4
  • Chromosomes, Human, Pair 8
  • Female
  • Genetic Testing*
  • Humans
  • In Situ Hybridization, Fluorescence
  • Intellectual Disability / genetics*
  • Karyotyping
  • Pedigree
  • Phenotype