Research Paper Volume 6, Issue 3 pp 231—245
Oxidative stress and antioxidant response in fibroblasts from Werner and Atypical Werner Syndromes
- 1 CIBERER. Centro de Investigación Biomédica en Red de Enfermedades Raras, Valencia, Spain
- 2 FIHCUV-INCLIVA. Valencia, Spain
- 3 Department of Physiology, Medicine School, University of Valencia, Valencia. Spain
- 4 Sistemas genómicos, Valencia, Spain
- 5 University of Valencia-UCIM, Spain
- 6 Universidad de Las Villas, Las Villas, Cuba
Received: January 23, 2014 Accepted: March 29, 2014 Published: March 31, 2014
https://doi.org/10.18632/aging.100649How to Cite
Abstract
Werner Syndrome (WS, ICD-10 E34.8, ORPHA902) and Atypical Werner Syndrome (AWS, ICD-10 E34.8, ORPHA79474) are very rare inherited syndromes characterized by premature aging. While approximately 90% of WS individuals have any of a range of mutations in the WRN gene, there exists a clinical subgroup in which the mutation occurs in the LMNA/C gene in heterozygosity. Although both syndromes exhibit an age-related pleiotropic phenotype, AWS manifests the onset of the disease during childhood, while major symptoms in WS appear between the ages of 20 and 30. To study the molecular mechanisms of progeroid diseases provides a useful insight into the normal aging process. Main changes found were the decrease in Cu/Zn and Mn SOD activities in the three cell lines. In AWS, both mRNA SOD and protein levels were also decreased. Catalase and glutathione peroxidases decrease, mainly in AWS. Glutaredoxin (Grx) and thioredoxin (Trx) protein expression was lower in the three progeroid cell lines. Grx and Trx were subjected to post-transcriptional regulation, because protein expression was reduced although mRNA levels were not greatly affected in WS.