RRC ID |
12081
|
著者 |
Taylor MR, Kikkawa S, Diez-Juan A, Ramamurthy V, Kawakami K, Carmeliet P, Brockerhoff SE.
|
タイトル |
The zebrafish pob gene encodes a novel protein required for survival of red cone photoreceptor cells.
|
ジャーナル |
Genetics
|
Abstract |
The zebrafish mutant, partial optokinetic response b (pob), was isolated using an N-ethyl N-nitrosourea (ENU)-based screening strategy designed to identify larvae with defective optokinetic responses in red but not white light. Previous studies showed that red-light blindness in pob is due to the specific loss of long-wavelength photoreceptor cells via an apoptotic mechanism. Here, we used positional cloning to identify the mutated pob gene. We find that pob encodes a highly conserved 30-kDa protein of unknown function. To demonstrate that the correct gene was isolated, we used the Tol2 transposon system to generate transgenic animals and rescue the mutant phenotype. The Pob protein contains putative transmembrane regions and protein-sorting signals. It is localized to the inner segment and synapse in photoreceptor cells, and when expressed in COS-7 cells it localizes to intracellular compartments. We also show that the degeneration of red cone photoreceptors in the mutants occurs independently of light. On the basis of our findings, we propose that Pob is not involved in phototransduction but rather plays an essential role in protein sorting and/or trafficking.
|
巻・号 |
170(1)
|
ページ |
263-73
|
公開日 |
2005-5-1
|
DOI |
10.1534/genetics.104.036434
|
PII |
genetics.104.036434
|
PMID |
15716502
|
PMC |
PMC1449739
|
MeSH |
Amino Acid Sequence
Animals
COS Cells
Cell Survival / genetics
Chlorocebus aethiops
Humans
Immunohistochemistry
In Situ Hybridization
Membrane Proteins / genetics*
Membrane Proteins / physiology
Mice
Molecular Sequence Data
Retinal Cone Photoreceptor Cells / metabolism*
Zebrafish / genetics*
Zebrafish / metabolism
Zebrafish Proteins / genetics*
Zebrafish Proteins / physiology
|
IF |
4.015
|
引用数 |
26
|
WOS 分野
|
GENETICS & HEREDITY
|
リソース情報 |
ゼブラフィッシュ |
Method |