Defective adgra2 (gpr124) splicing and function in zebrafish ouchless mutants

Author:

Bostaille Naguissa1,Gauquier Anne1,Stainier Didier Y. R.2,Raible David W.3,Vanhollebeke Benoit14ORCID

Affiliation:

1. Laboratory of Neurovascular Signaling, Department of Molecular Biology, ULB Neuroscience Institute, Université libre de Bruxelles (ULB), B-6041 Gosselies, Belgium

2. Department of Developmental Genetics, Max Planck Institute for Heart and Lung Research, 61231 Bad Nauheim, Germany

3. Department of Biological Structure, University of Washington, Seattle, WA 98195, USA

4. Center for Microscopy and Molecular Imaging, Université libre de Bruxelles (ULB), B-6041 Gosselies, Belgium

Abstract

ABSTRACT A hitherto unidentified N-ethyl-N-nitrosourea (ENU)-induced mutation affects dorsal root ganglia (DRG) formation in ouchless mutant zebrafish larvae. In contrast to previous findings assigning the ouchless phenotypes to downregulated sorbs3 transcript levels, this work re-attributes the phenotypes to an essential splice site mutation affecting adgra2 (gpr124) splicing and function. Accordingly, ouchless mutants fail to complement previously characterized adgra2 mutants and exhibit highly penetrant cerebrovascular defects. The aberrantly spliced adgra2 transcript found in ouchless mutants encodes a receptor lacking a single leucine-rich repeat (LRR) within its N-terminus.

Funder

Fonds de la Recherche Scientifique - FNRS

Fondation Université libre de Bruxelles

European Regional Development Fund

Fédération Wallonie-Bruxelles

Publisher

The Company of Biologists

Subject

Developmental Biology,Molecular Biology

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