Evaluating drug targets through human loss-of-function genetic variation

EV Minikel, KJ Karczewski, HC Martin, BB Cummings… - Nature, 2020 - nature.com
Naturally occurring human genetic variants that are predicted to inactivate protein-coding
genes provide an in vivo model of human gene inactivation that complements knockout …

Identification of cis-suppression of human disease mutations by comparative genomics

DM Jordan, SG Frangakis, C Golzio, CA Cassa… - Nature, 2015 - nature.com
Patterns of amino acid conservation have served as a tool for understanding protein
evolution. The same principles have also found broad application in human genomics …

The mutational constraint spectrum quantified from variation in 141,456 humans

KJ Karczewski, LC Francioli, G Tiao, BB Cummings… - Nature, 2020 - nature.com
Genetic variants that inactivate protein-coding genes are a powerful source of information
about the phenotypic consequences of gene disruption: genes that are crucial for the …

From target discovery to clinical drug development with human genetics

K Trajanoska, C Bhérer, D Taliun, S Zhou, JB Richards… - Nature, 2023 - nature.com
The substantial investments in human genetics and genomics made over the past three
decades were anticipated to result in many innovative therapies. Here we investigate the …

Genetic associations of protein-coding variants in human disease

BB Sun, MI Kurki, CN Foley, A Mechakra, CY Chen… - Nature, 2022 - nature.com
Genome-wide association studies (GWAS) have identified thousands of genetic variants
linked to the risk of human disease. However, GWAS have so far remained largely …

The support of human genetic evidence for approved drug indications

MR Nelson, H Tipney, JL Painter, J Shen, P Nicoletti… - Nature …, 2015 - nature.com
Over a quarter of drugs that enter clinical development fail because they are ineffective.
Growing insight into genes that influence human disease may affect how drug targets and …

The landscape of tolerated genetic variation in humans and primates

H Gao, T Hamp, J Ede, JG Schraiber, J McRae… - Science, 2023 - science.org
Personalized genome sequencing has revealed millions of genetic differences between
individuals, but our understanding of their clinical relevance remains largely incomplete. To …

Protein-altering variants at copy number-variable regions influence diverse human phenotypes

MLA Hujoel, RE Handsaker, MA Sherman… - Nature Genetics, 2024 - nature.com
Copy number variants (CNVs) are among the largest genetic variants, yet CNVs have not
been effectively ascertained in most genetic association studies. Here we ascertained …

Using human genetics to improve safety assessment of therapeutics

KJ Carss, AM Deaton, A Del Rio-Espinola… - Nature Reviews Drug …, 2023 - nature.com
Human genetics research has discovered thousands of proteins associated with complex
and rare diseases. Genome-wide association studies (GWAS) and studies of Mendelian …

Validating therapeutic targets through human genetics

RM Plenge, EM Scolnick, D Altshuler - Nature reviews Drug discovery, 2013 - nature.com
More than 90% of the compounds that enter clinical trials fail to demonstrate sufficient safety
and efficacy to gain regulatory approval. Most of this failure is due to the limited predictive …