Familial hypercholesterolaemia (FH) is an autosomal dominant genetic disorder characterised by severe hypercholesterolaemia leading to premature coronary artery disease (CAD). Oxidised low-density lipoprotein (ox-LDL), F2-isoprostanes (ISP) and malondialdehyde (MDA) are established oxidative stress biomarker, but the status of oxidative stress in FH is not well studied. The aim of this study is to investigate oxidative stress status among FH patients and normocholesterolaemic control (NC) subjects. Ninety-eight FH patients and 100 (age, gender and BMI matched) NC subjects were recruited in series of health screening programmes across the country. Fasting blood samples were analysed for serum ox-LDL, ISP and MDA. Ox-LDL, ISP and MDA concentrations were higher in FH groups compared to NC (mean±SEM: 63.0±6.5 vs 25.5±1.2 (U/l), p<0.001); 749.7±74.0 vs 354.2±18.1 pg/ml, p<0.0001; 342.4±46.0 vs 162.7±13.5 nmol/g, p<0.0001). Ox-LDL showed correlation with glucose (p<0.05), TC (p<0.001), LDL-c (p<0.001) and HDL-c (p<0.01) in all subjects. LDL-c was associated positively with ox-LDL concentration (p<0.001). LDL-c was an independent predictor for ox-LDL concentration (p<0.001) after adjustment for the various confounding factors. In conclusion, FH patients have higher oxidative stress status which contributes to the greater risk of developing atherosclerosis and its related complications. LDL concentration is an independent determinant of ox-LDL, suggesting that both the proatherogenic quantity and quality of LDL coexist in FH which enhance the risk of premature CAD.