BACKGROUND AND AIMS: Familial hypercholesterolemia (FH) is a genetic disorder characterized by elevated low-density lipoprotein cholesterol (LDL-C) and increased risk of premature coronary heart disease (CHD). While current LDL-C levels usually guides therapy, the cumulative exposure to LDL-C (the LDL-C burden) is suggested to offer a more precise estimate of cardiovascular risk in people with FH. Therefore, using real-world data, this study aimed to estimate the LDL-C burden at different ages in elderly FH patients with and without CHD, and to assess the LDL-C burden at CHD onset. METHODS: Data was retrospectively collected from the medical records of elderly (>60 years) FH patients at the Lipid Clinic in Oslo. The LDL-C burden (mM-years) was estimated based on repeated LDL-C measurements and information on lipid-lowering medication. Time-weighted average (TWA) LDL-C was calculated as LDL-C burden divided by years. RESULTS: We included 112 FH patients, of which 55 (49 %) had experienced at least one CHD-event, and 58 (52 %) were females. Median age at first and last visit were 50 years and 68 years, respectively, with a median of 9 (range; 2-14) available LDL-C measurements. Subjects with CHD had higher LDL-C burden at all ages tested (45, 50 and 60 years) compared with the non-CHD group ( < 0.01, also after adjusting for sex), and had higher TWA LDL-C before treatment at the Lipid Clinic ( = 0.004), but not during follow-up ( = 0.6). There were no sex differences in LDL-C burden at all ages tested, also after adjusting for CHD ( > 0.1). However, women had higher TWA LDL-C during follow-up at the Lipid Clinic ( = 0.01). Median LDL-C burden at CHD onset was 352 mM-years; numerically lower in women than in men (320 vs. 357 mM-years, respectively. = 0.1). CONCLUSION: Elderly FH patients with CHD had higher estimated LDL-C burden compared with FH patients without CHD, due to higher burden , highlighting the importance of earlydetection and treatment.