DOI work
Fetched
Post
crossref
Hellen Weinschutz Mendes, Uma Neelakantan, Yunqing Liu, Sarah E. Fitzpatrick, Tianying Chen, Weimiao Wu, April Pruitt, David S. Jin, et al.
(2023).
High-throughput functional analysis of autism genes in zebrafish identifies convergence in dopaminergic and neuroimmune pathways.
Cell Reports, 42(3), 112243.
Elsevier BV.
- Publication date
-
March 2023
- Identifier
-
10.1016/j.celrep.2023.112243
- Authors
-
Hellen Weinschutz Mendes,
Uma Neelakantan,
Yunqing Liu,
Sarah E. Fitzpatrick,
Tianying Chen,
Weimiao Wu,
April Pruitt,
David S. Jin,
Priyanka Jamadagni,
Marina Carlson,
Cheryl M. Lacadie,
Kristen D. Enriquez,
Ningshan Li,
Dejian Zhao,
Sundas Ijaz,
Catalina Sakai,
Christina Szi,
Brendan Rooney,
Marcus Ghosh,
Ijeoma Nwabudike,
Andrea Gorodezky,
Sumedha Chowdhury,
Meeraal Zaheer,
Sarah McLaughlin,
Joseph M. Fernandez,
Jia Wu,
Jeffrey A. Eilbott,
Brent Vander Wyk,
Jason Rihel,
Xenophon Papademetris,
Zuoheng Wang,
Ellen J. Hoffman
- Source
- Cell Reports
- Details
- 42(3), 112243
- Reference type
- article
- Publisher
- Elsevier BV
- Metadata source
- crossref
Abstract
Advancing from gene discovery in autism spectrum disorders (ASDs) to the identification of biologically relevant mechanisms remains a central challenge. Here, we perform parallel in vivo functional analysis of 10 ASD genes at the behavioral, structural, and circuit levels in zebrafish mutants, revealing both unique and overlapping effects of gene loss of function. Whole-brain mapping identifies the forebrain and cerebellum as the most significant contributors to brain size differences, while regions involved in sensory-motor control, particularly dopaminergic regions, are associated with altered baseline brain activity. Finally, we show a global increase in microglia resulting from ASD gene loss of function in select mutants, implicating neuroimmune dysfunction as a key pathway relevant to ASD biology.
Study authors in this cited reference