First report of a short in-frame biallelic deletion removing part of the EGF-like domain calcium-binding motif in LTBP4 and causing autosomal recessive cutis laxa type 1C.
Jean-Marie RavelMargot ComelMarion WandzelMyriam BronnerAurélie TatopoulosMathilde RenaudLaëtitia LambertAnne-Claire BursztejnCéline BonnetPublished in: American journal of medical genetics. Part A (2022)
Cutis laxa (CL) is a rare connective tissue disorder characterized by wrinkled, abundant and sagging skin, sometimes associated with systemic impairment. Biallelic alterations in latent transforming growth factor beta-binding protein 4 gene (LTBP4) cause autosomal recessive type 1C cutis laxa (ARCL1C, MIM #613177). The present report describes the case of a 17-months-old girl with cutis laxa together with a literature review of previous ARCL1C cases. Based on proband main clinical signs (cutis laxa and pulmonary emphysema), clinical exome sequencing (CES) was performed and showed a new nine base-pairs homozygous in-frame deletion in LTBP4 gene. RT-PCR and cDNA Sanger sequencing were performed in order to clarify its impact on RNA. This report demonstrates that a genetic alteration in the EGF-like 14 domain calcium-binding motif of LTBP4 gene is likely responsible for cutis laxa in our patient.
Keyphrases
- copy number
- transforming growth factor
- intellectual disability
- genome wide
- binding protein
- case report
- epithelial mesenchymal transition
- single cell
- genome wide identification
- dna methylation
- growth factor
- chronic obstructive pulmonary disease
- autism spectrum disorder
- gene expression
- dna binding
- lung function
- signaling pathway
- cystic fibrosis
- idiopathic pulmonary fibrosis
- muscular dystrophy
- soft tissue
- duchenne muscular dystrophy