PUBLICATION
Interaction of Axonal Chondrolectin with Collagen XIXa1 Is Necessary for Precise Neuromuscular Junction Formation
- Authors
- Oprişoreanu, A.M., Smith, H.L., Arya, S., Webster, R., Zhong, Z., Wehner, D., Cardozo, M.J., Becker, T., Talbot, K., Becker, C.G.
- ID
- ZDB-PUB-191031-13
- Date
- 2019
- Source
- Cell Reports 29: 1082-1098.e10 (Journal)
- Registered Authors
- Arya, Sukrat, Becker, Catherina G., Becker, Thomas, Cardozo, Marcos, Smith, Hannah, Wehner, Daniel
- Keywords
- CaP motor neurons, axon growth, extracellular matrix, horizontal myoseptum, muscle pioneer cells, neuromuscular junction, stalled motor axons, synapse, zebrafish
- MeSH Terms
-
- Animals
- Axons/metabolism*
- Conserved Sequence
- Electrophysiological Phenomena
- Escape Reaction
- Evolution, Molecular
- Fibril-Associated Collagens/metabolism*
- Gene Expression Regulation, Developmental
- HEK293 Cells
- Humans
- Larva/physiology
- Lectins, C-Type/chemistry
- Lectins, C-Type/genetics
- Lectins, C-Type/metabolism*
- Mice
- Motor Activity
- Motor Endplate/metabolism
- Motor Neurons/metabolism
- Mutation/genetics
- Neurites/metabolism
- Neurogenesis
- Neuromuscular Junction/growth & development*
- Neuromuscular Junction/metabolism*
- Phenotype
- Protein Binding
- Protein Domains
- Synapses/metabolism
- Zebrafish/metabolism*
- Zebrafish Proteins/chemistry
- Zebrafish Proteins/genetics
- Zebrafish Proteins/metabolism*
- PubMed
- 31665626 Full text @ Cell Rep.
Citation
Oprişoreanu, A.M., Smith, H.L., Arya, S., Webster, R., Zhong, Z., Wehner, D., Cardozo, M.J., Becker, T., Talbot, K., Becker, C.G. (2019) Interaction of Axonal Chondrolectin with Collagen XIXa1 Is Necessary for Precise Neuromuscular Junction Formation. Cell Reports. 29:1082-1098.e10.
Abstract
Chondrolectin (Chodl) is needed for motor axon extension in zebrafish and is dysregulated in mouse models of spinal muscular atrophy (SMA). However, the mechanistic basis of Chodl function is not known. Here, we use Chodl-deficient zebrafish and mouse mutants to show that the absence of Chodl leads to anatomical and functional defects of the neuromuscular synapse. In zebrafish, the growth of an identified motor axon beyond an "en passant" synapse and later axon branching from synaptic points are impaired, leading to functional deficits. Mechanistically, motor-neuron-autonomous Chodl function depends on its intracellular domain and on binding muscle-derived collagen XIXa1 by its extracellular C-type lectin domain. Our data support evolutionarily conserved roles of Chodl in synaptogenesis and provide evidence for a "synapse-first" scenario of motor axon growth in zebrafish.
Errata / Notes
This article is corrected by ZDB-PUB-220906-213 .
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping