PUBLICATION
A screen for mutations in zebrafish that affect myelin gene expression in Schwann cells and oligodendrocytes
- Authors
- Kazakova, N., Li, H., Mora, A., Jessen, K.R., Mirsky, R., Richardson, W.D., and Smith, H.K.
- ID
- ZDB-PUB-060724-4
- Date
- 2006
- Source
- Developmental Biology 297(1): 1-13 (Journal)
- Registered Authors
- Richardson, William D., Smith, Hazel K.
- Keywords
- Myelin, Zebrafish, Schwann cells, Oligodendrocytes, Genetic screen, Retinoic acid
- MeSH Terms
-
- Animals
- Carrier Proteins/genetics
- Embryo, Nonmammalian
- Female
- Gene Expression Regulation, Developmental
- Male
- Mutation*
- Myelin Basic Protein/genetics
- Myelin Sheath/genetics*
- Nervous System/pathology
- Neuroglia/pathology
- Oligodendroglia/pathology
- Oligodendroglia/physiology*
- Schwann Cells/pathology
- Schwann Cells/physiology*
- Tretinoin/metabolism
- Tretinoin/pharmacology
- Zebrafish/embryology
- Zebrafish/genetics*
- Zebrafish/growth & development
- Zebrafish Proteins/drug effects
- Zebrafish Proteins/genetics
- p-Aminoazobenzene/analogs & derivatives
- p-Aminoazobenzene/pharmacology
- PubMed
- 16839543 Full text @ Dev. Biol.
- CTD
- 16839543
Citation
Kazakova, N., Li, H., Mora, A., Jessen, K.R., Mirsky, R., Richardson, W.D., and Smith, H.K. (2006) A screen for mutations in zebrafish that affect myelin gene expression in Schwann cells and oligodendrocytes. Developmental Biology. 297(1):1-13.
Abstract
Myelin is the multi-layered glial sheath around axons in the vertebrate nervous system. Myelinating glia develop and function in intimate association with neurons and neuron-glial interactions control much of the life history of these cells. However, many of the factors that regulate key aspects of myelin development and maintenance remain unknown. To discover new molecules that are important for glial development and myelination, we undertook a screen of zebrafish mutants with previously characterized neural defects. We screened for myelin basic protein (mbp) mRNA by in situ hybridization and identified four mutants (neckless, motionless, iguana and doc) that lacked mbp expression in parts of the peripheral and central nervous systems (PNS or CNS), despite the presence of axons. In all four mutants electron microscopy revealed that myelin-forming glia were present and had formed loose wraps around axons but did not form compact myelin. We found that addition of exogenous retinoic acid (RA) rescued mbp expression in neckless mutant embryos, which lack endogenous RA synthesis. Timed application of the RA synthesis inhibitor DEAB to wild type embryos showed that RA signalling is required at least 48 h before the onset of myelin protein synthesis in both CNS and PNS.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping