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PMID:20692250

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Citation

Larson, TA, Gordon, TN, Lau, HE and Parichy, DM (2010) Defective adult oligodendrocyte and Schwann cell development, pigment pattern, and craniofacial morphology in puma mutant zebrafish having an alpha tubulin mutation. Dev. Biol. 346:296-309

Abstract

The processes of myelination remain incompletely understood but are of profound biomedical importance owing to the several dysmyelinating and demyelinating disorders known in humans. Here, we analyze the zebrafish puma mutant, isolated originally for pigment pattern defects limited to the adult stage. We show that puma mutants also have late-arising defects in Schwann cells of the peripheral nervous system, locomotor abnormalities, and sex-biased defects in adult craniofacial morphology. Using methods of positional cloning, we identify a critical genetic interval harboring two alpha tubulin loci, and we identify a chemically induced missense mutation in one of these, tubulin alpha 8-like 3a (tuba8l3a). We demonstrate tuba8l3a expression in the central nervous system (CNS), leading us to search for defects in the development of oligodendrocytes, the myelinating cells of the CNS. We find gross reductions in CNS myelin and oligodendrocyte numbers in adult puma mutants, and these deficits are apparent already during the larval-to-adult transformation. By contrast, analyses of embryos and early larvae reveal a normal complement of oligodendrocytes that nevertheless fail to localize normal amounts of myelin basic protein (mbp) mRNA in cellular processes, and fail to organize these processes as in the wild-type. This study identifies the puma mutant as a valuable model for studying microtubule-dependent events of myelination, as well as strategies for remyelination in the adult.

Links

PubMed PMC2957186 Online version:10.1016/j.ydbio.2010.07.035

Keywords

Animals; Apoptosis Regulatory Proteins/genetics; Apoptosis Regulatory Proteins/metabolism; Base Sequence; Central Nervous System/embryology; Central Nervous System/metabolism; Embryo, Nonmammalian/metabolism; Molecular Sequence Data; Mutation; Oligodendroglia/cytology; Oligodendroglia/metabolism; Phylogeny; Proto-Oncogene Proteins/genetics; Proto-Oncogene Proteins/metabolism; RNA, Messenger/metabolism; Schwann Cells/cytology; Schwann Cells/metabolism; Skin Pigmentation/genetics; Tubulin/genetics; Zebrafish/embryology; Zebrafish/genetics; Zebrafish Proteins/genetics; Zebrafish Proteins/metabolism

Significance

Annotations

Gene product Qualifier GO Term Evidence Code with/from Aspect Extension Notes Status

DANRE:Q6DC49

involved_in

GO:0021782: glial cell development

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-GENO-101025-1

P

Seeded From UniProt

complete

DANRE:Q6DC49

involved_in

GO:0042552: myelination

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-GENO-101025-1

P

Seeded From UniProt

complete


See also

References

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