PUBLICATION
Zebrafish Class 1 Phosphatidylinositol Transfer Proteins: PITPbeta and Double Cone Cell Outer Segment Integrity in Retina
- Authors
- Ile, K.E., Kassen, S., Cao, C., Vihtehlic, T., Shah, S.D., Mousley, C.J., Alb, J.G. Jr, Huijbregts, R.P., Stearns, G.W., Brockerhoff, S.E., Hyde, D.R., and Bankaitis, V.A.
- ID
- ZDB-PUB-100621-10
- Date
- 2010
- Source
- Traffic (Copenhagen, Denmark) 11(9): 1151-1167 (Journal)
- Registered Authors
- Brockerhoff, Susan, Hyde, David R., Kassen, Sean, Stearns, George
- Keywords
- zebrafish, lipid signaling, retinal development, cone cells
- MeSH Terms
-
- Animals
- Models, Animal
- Phospholipid Transfer Proteins/chemistry
- Phospholipid Transfer Proteins/genetics
- Phospholipid Transfer Proteins/metabolism*
- Phospholipid Transfer Proteins/physiology
- Protein Isoforms
- Retinal Photoreceptor Cell Outer Segment/metabolism*
- Saccharomyces cerevisiae/genetics
- Zebrafish*
- PubMed
- 20545905 Full text @ Traffic
Citation
Ile, K.E., Kassen, S., Cao, C., Vihtehlic, T., Shah, S.D., Mousley, C.J., Alb, J.G. Jr, Huijbregts, R.P., Stearns, G.W., Brockerhoff, S.E., Hyde, D.R., and Bankaitis, V.A. (2010) Zebrafish Class 1 Phosphatidylinositol Transfer Proteins: PITPbeta and Double Cone Cell Outer Segment Integrity in Retina. Traffic (Copenhagen, Denmark). 11(9):1151-1167.
Abstract
Phosphatidylinositol transfer proteins (PITPs) in yeast coordinate lipid metabolism with the activities of specific membrane trafficking pathways. The structurally unrelated metazoan-specific PITPs (mPITPs), on the other hand, are an under-investigated class of proteins. It remains unclear what biological activities mPITPs discharge, and the mechanisms by which these proteins function are also not understood. The soluble class 1 mPITPs include the PITPalpha and PITPbeta isoforms. Of these, the beta-isoforms are particularly poorly characterized. Herein, we report the use of zebrafish as a model vertebrate for the study of class 1 mPITP biological function. Zebrafish express PITPalpha and PITPbeta-isoforms (Pitpna and Pitpnb, respectively) and a novel PITPbeta-like isoform (Pitpng). Pitpnb expression is particularly robust in double cone cells of the zebrafish retina. Morpholino-mediated protein knockdown experiments demonstrate Pitpnb activity is primarily required for biogenesis/maintenance of the double cone photoreceptor cell outer segments in the developing retina. By contrast, Pitpna activity is essential for successful navigation of early developmental programs. This study reports the initial description of the zebrafish class 1 mPITP family, and the first analysis of PITPbeta function in a vertebrate.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping