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Article

Zebrafish knockout models of atxn1a, atxn1b, and atxn1l reveal distinct and shared phenotypic and transcriptomic alterations.

Human molecular genetics - 10 Aug 2026

Karim Anwarul, Keerthisinghe Pramuk, Sarasamma Sreeja, Ciaburri Nicholas A, Giraldez Mabel Guerra, Naidoo Kylan, Orengo James P

Abstract excerpt

Spinocerebellar ataxia type 1 is a progressive neurodegenerative disorder caused by polyglutamine expansion in ATXN1, yet the normal physiological roles of ATXN1 and its paralog ATXN1L remain incompletely understood. To define these roles, we generated the first zebrafish knockouts (KOs) of the three ataxin-1 family genes, atxn1a, atxn1b, and atxn1l, using CRISPR/Cas9 and performed phenotypic and transcriptomic...

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Methodology Data sources Editorial policy Privacy About Contact Helena Bioinformatics

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Identifiers

PMID: 42611733
DOI: 10.1093/hmg/ddag078

Genes

ATXN1
ATXN1L

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PubMed metadata and Literature Corpus relationships. Corpus snapshot updated 8 Sept 2026.

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