Article
USP15 mutation associated with autism spectrum disorder alters progenitor fate and neuronal maturation in human brain organoids.
Molecules and cells - 1 Sept 2026
Park Tae-Hwan, Koh In Gyeong, Sung Seoyoung, Park Hayoon, Kim Jieun, Lee Sebin, Lee Yun Jin, Ko Hyunsoo, Han Jae Hyun, Bong Guiyoung, Yoo Hee Jeong, Kim Jaesang, An Joon-Yong, Lee Ji Yeoun
Abstract excerpt
De novo variants in the ubiquitin-proteasome pathway are linked to autism spectrum disorder (ASD), yet their functional impact on neurodevelopment remains poorly understood. We investigated USP15, a deubiquitinating enzyme with rare damaging variants identified in individuals with ASD, using isogenic human iPSC-derived brain organoids and single-cell transcriptomics. USP15 mutant organoids showed...
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