[Analysis of pathogenic variants in a Chinese pedigree affected with hyaline fibromatosis syndrome]

Zhonghua Yi Xue Yi Chuan Xue Za Zhi. 2021 Mar 10;38(3):232-237. doi: 10.3760/cma.j.cn511374-20200625-00472.
[Article in Chinese]

Abstract

Objective: To explore the clinical characteristics and genetic basis for a pair of twins affected with hyaline fibromatosis syndrome (HFS).

Methods: Clinical data of the twins were retrospectively analyzed. High-throughput sequencing was carried out to detect potential pathogenic variants. CLUSTALX was employed to analyze cross-species conservation of the mutant amino acids. Impact of the mutations was predicted by using software including PolyPhen-2 and Mutation taster.

Results: The pair of twins have featured growth and intelligence retardation, and were found to carry compound heterozygous variants of the ANTXR2 gene including c.1214G>A and c.1074delT, among which c.1214G>A was unreported previously. Both variants were predicted to be pathogenic. In addition to growth and mental delay, the pair of twins also featured hyperplasia of the gum and soft tissue-like masses of the auricle. The younger brother had rupture of the auricle mass during follow-up.

Conclusion: The patients' condition can probably be attributed to the compound heterozygous variants of the ANTXR2 gene. Above finding has facilitated molecular diagnosis of the patients.

Publication types

  • Case Reports

MeSH terms

  • Asian People / genetics
  • China
  • Humans
  • Hyaline Fibromatosis Syndrome* / genetics
  • Male
  • Mutation
  • Pedigree
  • Receptors, Peptide* / genetics
  • Retrospective Studies

Substances

  • ANTXR2 protein, human
  • Receptors, Peptide