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Updated: Mar 1, 2026

A Familial Hypercholesterolemia Human Liver Chimeric Mouse Model Using Induced Pluripotent Stem Cell-derived Hepatocytes
Published on: September 15, 2018
Genetic determinants of inherited susceptibility to hypercholesterolemia - a comprehensive literature review
C S Paththinige1, N D Sirisena2, Vhw Dissanayake2
1Human Genetics Unit, Faculty of Medicine, University of Colombo, Kynsey Road, Colombo, 00800, Sri Lanka. paththinige@yahoo.com.
Insights
Genetic variants beyond the four known familial hypercholesterolemia genes contribute to high cholesterol. Research identifies numerous genes and non-coding variants influencing lipid levels, suggesting polygenic inheritance and new therapeutic targets.
Area of Science:
- Genetics and Molecular Biology
- Cardiovascular Disease Research
- Metabolic Disorders
Background:
- Hypercholesterolemia significantly increases cardiovascular disease risk and mortality.
- While mutations in LDLR, APOB, PCSK9, and LDLRAP1 explain many familial cases, a substantial portion of hypercholesterolemia remains genetically unexplained.
- This suggests a polygenic basis involving numerous genes and regulatory elements.
Purpose of the Study:
- To review current evidence on genetic variants associated with hypercholesterolemia and key lipid traits (total cholesterol, HDL-C, LDL-C).
- To explore the biological pathways and pathogenetic mechanisms implicated by these genetic associations.
- To highlight the potential for discovering novel therapeutic and diagnostic strategies.
Main Methods:
- Systematic review of genome-wide association studies (GWAS) and candidate gene studies.
- Identification of single nucleotide variants (SNVs) linked to hypercholesterolemia and lipid levels.
- Analysis of gene functions related to lipid metabolism and non-metabolic pathways.
Main Results:
- Nearly 80 genes involved in lipid metabolism show associated SNVs.
- An additional ~120 genes, not previously known for lipid involvement, also have associated SNVs.
- Over 90% of identified SNVs are located in non-coding regions, suggesting regulatory roles.
Conclusions:
- A significant number of unrecognized genetic factors and regulatory mechanisms contribute to lipid homeostasis and hypercholesterolemia.
- Understanding these pathways is crucial for developing novel treatments and preventive strategies.
- Identification of molecular markers can improve risk prediction and early detection of hypercholesterolemia.
Abstract:
Hypercholesterolemia is a strong determinant of mortality and morbidity associated with cardiovascular diseases and a major contributor to the global disease burden. Mutations in four genes (LDLR, APOB, PCSK9 and LDLRAP1) account for the majority of cases with familial hypercholesterolemia. However, a substantial proportion of adults with hypercholesterolemia do not have a mutation in any of these four genes. This indicates the probability of having other genes with a causative or contributory role in the pathogenesis of hypercholesterolemia and suggests a polygenic inheritance of this condition. Here in, we review the recent evidence of association of the genetic variants with hypercholesterolemia and the three lipid traits; total cholesterol (TC), HDL-cholesterol (HDL-C) and LDL-cholesterol (LDL-C), their biological pathways and the associated pathogenetic mechanisms. Nearly 80 genes involved in lipid metabolism (encoding structural components of lipoproteins, lipoprotein receptors and related proteins, enzymes, lipid transporters, lipid transfer proteins, and activators or inhibitors of protein function and gene transcription) with single nucleotide variants (SNVs) that are recognized to be associated with hypercholesterolemia and serum lipid traits in genome-wide association studies and candidate gene studies were identified. In addition, genome-wide association studies in different populations have identified SNVs associated with TC, HDL-C and LDL-C in nearly 120 genes within or in the vicinity of the genes that are not known to be involved in lipid metabolism. Over 90% of the SNVs in both these groups are located outside the coding regions of the genes. These findings indicates that there might be a considerable number of unrecognized processes and mechanisms of lipid homeostasis, which when disrupted, would lead to hypercholesterolemia. Knowledge of these molecular pathways will enable the discovery of novel treatment and preventive methods as well as identify the biochemical and molecular markers for the risk prediction and early detection of this common, yet potentially debilitating condition.
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