Detection of regions of homozygosity in an unusual case of frontonasal dysplasia

Abstract We present the case of a 7-year-old Ecuadorian mestizo girl with multiple orofacial malformations. The patient is the product of a first-degree relationship (father–daughter). A cytogenetic study revealed a normal karyotype. The genetic mapping array study identified 0.73 Gb of alterations,...

Full description

Saved in:
Bibliographic Details
Main Authors: César Paz-y-Miño, Ramón Miguel Vargas-Vera, Martha Verónica Placencia-Ibadango, Kalid Stefano Vargas-Silva, Juan Luis García-Hernández, Thalía Balarezo-Díaz, Paola E. Leone
Format: Article
Language:English
Published: BMC 2024-10-01
Series:Molecular Cytogenetics
Subjects:
Online Access:https://doi.org/10.1186/s13039-024-00693-1
Tags: Add Tag
No Tags, Be the first to tag this record!
Description
Summary:Abstract We present the case of a 7-year-old Ecuadorian mestizo girl with multiple orofacial malformations. The patient is the product of a first-degree relationship (father–daughter). A cytogenetic study revealed a normal karyotype. The genetic mapping array study identified 0.73 Gb of alterations, 727,087,295 bp involved in regions of homozygosity (ROH) in all chromosomes (25.2% of the genome) and 764,028 bp in gains in chromosomes 9 and 14. Genes from the TGFB, BMP, FGF, SHH and WNT families, among others, were identified in the ROH. They are related to craniofacial development and their protein products showed a strong association in the interactome analysis.
ISSN:1755-8166