a Laboratory of Neurological Disease Modeling and Translational Research, West China Hospital, Sichuan University, No. 37 Guoxue Alley, Wuhou District, Chengdu 610041, China;
b Medical Innovation Center, Sichuan University of Science & Engineering, Zigong, 643002, China;
c Key Laboratory of Drug Targeting and Drug Delivery System of the Education Ministry and Sichuan Province, Sichuan Engineering Laboratory for Plant-Sourced Drug and Sichuan Research Center for Drug Precision Industrial Technology, West China School of Pharmacy, Sichuan University, Chengdu 610041, China;
d Medical College of Tibet University, Lasa, 850002, China;
e Tianfu Jincheng Laboratory, City of Future Medicine, Chengdu, 641400, China
Funds:
This project was supported by 1. National Natural Science Foundation of China (82071349)
2. Sichuan Science and Technology Program (2025ZNSFSC0703)
3. Young Scientists Fund of the National Natural Science Foundation of China (Grant No. 82204513)
4. Natural Science Foundation of Sichuan Province, China (Grant No. 2023NSFSC1673)
5. Innovation Guidance Foundation of the Key Laboratory of DrugTargeting and Drug Delivery System of the Education Ministry and Sichuan Province (Grant No. SCU2023D005)
6. Scientific Research Staring Foundation of Sichuan University (Grant No. YJ202165).
Clustered regularly interspaced short palindromic repeats (CRISPR)-Cas9 screening technology is redefining the landscape of drug discovery and therapeutic target identification by providing a precise and scalable platform for functional genomics. The development of extensive single-guide RNA (sgRNA) libraries enables high-throughput screening (HTS) that systematically investigates gene-drug interactions across the genome. This powerful approach has found broad applications in identifying drug targets for various diseases, including cancer, infectious diseases, metabolic disorders, and neurodegenerative conditions, playing a crucial role in elucidating drug mechanisms and facilitating drug screening. Despite challenges like off-target effects, data complexity, and ethical or regulatory concerns, ongoing advancements in CRISPR technology and bioinformatics are steadily overcoming these limitations. Additionally, by integrating with organoid models, artificial intelligence (AI), and big data technologies, CRISPR screening expands the scale, intelligence, and automation of drug discovery. This integration boosts data analysis efficiency and offers robust support for uncovering new therapeutic targets and mechanisms. This review outlines the fundamental principles and applications of CRISPR screening technology, delves into specific case studies and technical challenges, and highlights its expanding role in drug discovery and target identification. It also examines the potential for clinical translation and addresses the associated ethical and regulatory considerations.