Language

English

Publication Date

6-1-2023

Journal

3 Biotech

DOI

10.1007/s13205-023-03535-w

PMID

37162806

PMCID

PMC10163994

PubMedCentral® Posted Date

5-6-2023

PubMedCentral® Full Text Version

Post-print

Abstract

Inherited retinal dystrophies (IRDs) include a large chronic heterogeneity genetic disease. While many disease-causing pathogenic variants were involved in the progression of IRD, the Ceramide Kinase Like (CERKL) gene variant in Iranian patients is not well characterized. In this study, a consanguineous Iranian family with three generations was recruited whom presented with the clinical diagnosis of autosomal recessive IRD. By targeted next-generation sequencing (TGS) and Sanger sequencing, the proband was found to have a novel, pathological homozygous deletion variant c.560_568del (p.187_190del) of the CERKL gene (NM_001030311.2) that co-segregated with the disease in all affected family members. The Cerkl is highly expressed in the later four developmental retinal stages, playing a vital role in retina degeneration. Therefore, the identification of a novel, homozygous deletion CERKL variant c.560_568del (p.187_190del) in an IRD familial cohort descent provides insights into the molecular pathogenesis of IRD and facilitates genetic counseling and disease prediction.

Keywords

Retinal dystrophy, CERKL gene, Deletion variant, Targeted next-generation sequencing (NGS), Iranian

Published Open-Access

yes

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