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Loss of zebrafish ctnnd2b results in disorganised forebrain neuron clusters

View ORCID ProfileWolfgang Hofmeister, Raquel Vaz, Steven Edwards, Alfredo Dueñas Rey, Anna Lindstrand
doi: https://doi.org/10.1101/420828
Wolfgang Hofmeister
1Department of Molecular Medicine and Surgery, Karolinska Institutet, 171 76 Stockholm, Sweden
3Centre of Molecular Medicine, Karolinska Institutet, 171 76 Stockholm, Sweden
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  • ORCID record for Wolfgang Hofmeister
  • For correspondence: wolfgang.hofmeister@ki.se
Raquel Vaz
1Department of Molecular Medicine and Surgery, Karolinska Institutet, 171 76 Stockholm, Sweden
3Centre of Molecular Medicine, Karolinska Institutet, 171 76 Stockholm, Sweden
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Steven Edwards
4Department of Applied Physics, Science for Life Laboratory, Royal Institute of Technology, Stockholm, Sweden (SE)
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Alfredo Dueñas Rey
1Department of Molecular Medicine and Surgery, Karolinska Institutet, 171 76 Stockholm, Sweden
2Department of Clinical Genetics, Karolinska University Hospital, 171 76 Stockholm, Sweden
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Anna Lindstrand
1Department of Molecular Medicine and Surgery, Karolinska Institutet, 171 76 Stockholm, Sweden
2Department of Clinical Genetics, Karolinska University Hospital, 171 76 Stockholm, Sweden
3Centre of Molecular Medicine, Karolinska Institutet, 171 76 Stockholm, Sweden
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  • For correspondence: wolfgang.hofmeister@ki.se
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Abstract

Delta catenin (CTNND2) is an adhesive junction associated protein belonging to the family of p120ct catenins. It is located on the short arm of chromosome 5, a region deleted in Cri-du-chat syndrome. Heterozygous loss of CTNND2 function has been linked to autism, schizophrenia, and mild intellectual disability with or without dyslexia-like learning difficulties. To date, most functional studies have focused on homozygous loss of the gene, contradictory to the dominant effect of loss of a single allele observed in neurodevelopmental disorders. Here we show that heterozygous loss of ctnnd2b results in a disorganisation and imbalance of neuronal subtypes in forebrain specific regions. Using the zebrafish model, we show that CRISPR/Cas9-induced loss of ctnnd2b but not ctnnd2a results in an increase in isl1-expressing cells and a local reduction of GABA expressing neurons in the optic recess region of the embryonic zebrafish forebrain. Using time-lapse analysis, we found that the disorganised distribution of is1l-expressing forebrain neurons was not due to migration defects, but rather an increase in the number of isl1-GFP neurons in the optic recess region. Upon closer analysis, these neurons appear disorganised and show an altered morphology and orientation. Overall this data suggests that ctnnd2 may affect the differentiation cascade of neuronal subtypes in specific regions of the vertebrate brain.

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Posted September 18, 2018.
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Loss of zebrafish ctnnd2b results in disorganised forebrain neuron clusters
Wolfgang Hofmeister, Raquel Vaz, Steven Edwards, Alfredo Dueñas Rey, Anna Lindstrand
bioRxiv 420828; doi: https://doi.org/10.1101/420828
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Loss of zebrafish ctnnd2b results in disorganised forebrain neuron clusters
Wolfgang Hofmeister, Raquel Vaz, Steven Edwards, Alfredo Dueñas Rey, Anna Lindstrand
bioRxiv 420828; doi: https://doi.org/10.1101/420828

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