PUBLICATION
Genomic structure and embryonic expression of zebrafish lysyl hydroxylase 1 and lysyl hydroxylase 2
- Authors
- Schneider, V.A., and Granato, M.
- ID
- ZDB-PUB-061031-4
- Date
- 2007
- Source
- Matrix biology : journal of the International Society for Matrix Biology 26(1): 12-19 (Journal)
- Registered Authors
- Granato, Michael, Schneider, Valerie
- Keywords
- Zebrafish, Lysyl hydroxylase, lh, plod, Extracellular matrix, Collagen
- MeSH Terms
-
- Amino Acid Sequence
- Animals
- Cloning, Molecular
- Embryo, Nonmammalian/embryology
- Embryo, Nonmammalian/enzymology
- Embryo, Nonmammalian/metabolism*
- Exons/genetics
- Gastrula/metabolism
- Gene Expression Regulation, Developmental
- Gene Expression Regulation, Enzymologic
- Isoenzymes/genetics
- Mesoderm/metabolism
- Molecular Sequence Data
- Notochord/metabolism
- Procollagen-Lysine, 2-Oxoglutarate 5-Dioxygenase/genetics*
- Sequence Homology, Amino Acid
- Somites/metabolism
- Zebrafish/embryology
- Zebrafish/genetics*
- Zebrafish Proteins/genetics*
- PubMed
- 17056240 Full text @ Matrix Biol.
Citation
Schneider, V.A., and Granato, M. (2007) Genomic structure and embryonic expression of zebrafish lysyl hydroxylase 1 and lysyl hydroxylase 2. Matrix biology : journal of the International Society for Matrix Biology. 26(1):12-19.
Abstract
Collagen biosynthesis in both invertebrates and vertebrates is critically dependent upon the activity of lysyl hydroxylase (LH) enzymes. In humans, mutations in the genes encoding LH1 and LH2 have been shown to cause two distinct connective tissue disorders, Ehlers-Danlos (Type VIA) and Bruck syndromes. While the biochemical properties of these enzymes have been intensively studied, their embryonic patterns of expression and developmental roles remain unknown. We now present the cloning and analyses of the genes encoding LH1 and LH2 in the zebrafish, Danio rerio. We find these genes to be similarly organized to other vertebrate lh (plod) genes, including the presence of an alternatively spliced exon in lh2. We also examine the mRNA expression patterns of lh1 and lh2 during embryogenesis and find them to exhibit unique and dynamic patterns of expression. These results strongly suggest that LH enzymes are not merely housekeeping enzymes, but play distinct developmental roles. The identification of these genes in the zebrafish, a genetic model organism whose development is well characterized, now provides the basis for the establishment of the first animal models for both Ehlers-Danlos (Type VIA) and Bruck syndromes.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping