Novel Mutation With Literature Review WW Domain-Containing Oxidoreductase (WWOX) Gene
Ghassan Sukkar1,2, Razan M Alzahrani3,1, Bsaim A Altirkistani2
1Department of Pediatrics, Ministry of National Guard - Health Affairs, Jeddah, SAU.
Insights
Genetic mutations in the WW domain-containing oxidoreductase (WWOX) gene cause severe infant epileptic encephalopathy. Whole-exome sequencing identified a homozygous WWOX variant, highlighting its role in brain development.
Area of Science:
- Genetics
- Neuroscience
- Molecular Biology
Background:
- The WW domain-containing oxidoreductase (WWOX) gene, a known tumor suppressor, is implicated in various metabolic and neural developmental disorders.
- Genetic alterations in WWOX are associated with autosomal recessive developmental and epileptic encephalopathy, presenting in infancy with refractory seizures, hypotonia, and psychomotor impairment.
Observation:
- Whole-exome sequencing (WES) was utilized to analyze patient DNA.
- The study identified a homozygous variant (c.406A>G) in the WWOX gene (OMIM:605131).
- Prenatal WES data confirmed parental heterozygosity and the homozygous status of the variant in the patient.
Findings:
- The identified homozygous WWOX variant is directly linked to developmental and epileptic encephalopathy.
- This finding underscores the critical role of the WWOX gene in normal brain development.
Implications:
- WWOX gene mutations are associated with developmental delay and severe neurological conditions.
- WES is recommended as a primary diagnostic tool for challenging cases, especially in populations with high consanguinity rates.
Abstract:
Genetic alterations in the WW domain-containing oxidoreductase (WWOX) gene cause autosomal recessive developmental and epileptic encephalopathy, characterized by the onset of refractory seizures in infants, along with severe axial hypotonia and profoundly impaired psychomotor development. It has also been expanded to include metabolism and endocrine systems. Despite its function as a tumor suppressor gene, genetic alterations in WWOX have been found in several metabolic disorders and neural diseases related to brain development. Whole-exome sequencing (WES) was performed on the patient sample. Genomic DNA was fragmented, and the exons of known genes in the human genome, as well as the corresponding exon-intron boundaries,were enriched using Roche KAPA capture technology (KAPA hyperExome Library, WES identifying the homozygous variant c.406A>G in WWOX (OMIM:605131). This variant of WWOX was also observed in the prenatal WES data, indicating that both parents were heterozygous carriers and the detected variant was homozygous. This study highlighted the importance of the human WWOX gene in brain development and the association between WWOX gene mutations and developmental delay. We recommend performing WES as a primary screening before the final diagnosis, particularly in populations with high rates of consanguinity and in clinically challenging cases.
More Related Videos
09:37Navigating MARRVEL, a Web-Based Tool that Integrates Human Genomics and Model Organism Genetics Information
Published on: August 15, 2019
06:53Visualization of Mitochondrial Respiratory Function using Cytochrome C Oxidase / Succinate Dehydrogenase COX/SDH Double-labeling Histochemistry
Published on: November 23, 2011
Related Concept Videos
Canonical Wnt Signaling Pathway
Non-Canonical Wnt Signaling Pathways
Mutations in Microorganisms
Pleiotropy
Mismatch Repair
The Mutator Protein Family Plays a Key Role in DNA Mismatch Repair
The human genome has more than 3 billion base pairs of DNA per cell. Prior to cell division, that vast amount of genetic...
Mutations
