PUBLICATION

Early defects in photoreceptor outer segment morphogenesis in zebrafish ift57, ift88 and ift172 Intraflagellar Transport mutants

Authors

Sukumaran, S., and Perkins, B.D.

ID

ZDB-PUB-090116-17

Date

2009

Source

Vision Research 49(4): 479-489 (Journal)

Registered Authors

Perkins, Brian

Keywords

Retina, Photoreceptor, Intraflagellar transport, Connecting cilium, Outer segment

MeSH Terms

Animals
Morphogenesis/genetics
Adaptor Proteins, Signal Transducing/genetics*
Mutation
Time Factors
Microscopy, Electron, Transmission
Immunohistochemistry
Zebrafish/embryology*
Zebrafish/genetics*
Zebrafish Proteins/genetics*
Embryo, Nonmammalian/anatomy & histology
Retinal Photoreceptor Cell Outer Segment/ultrastructure*

(all 12)

PubMed

19136023 Full text @ Vision Res.

Abstract

Intraflagellar Transport (IFT) refers to a highly conserved process occurring in eukaryotic ciliated structures. In vertebrate photoreceptors, IFT mediates protein trafficking to the outer segments. The IFT particle is a multi-subunit complex and mutations in many individual components causes photoreceptor defects. In zebrafish, mutations in the ift57, ift88, and ift172 genes result in retinal degeneration by 5 days post fertilization (dpf). Although the effects of these mutations on photoreceptor survival have been described, early developmental morphogenesis remains poorly understood. We used transmission electron microscopy and immunohistochemistry to examine these mutants at 60, 72, and 96h post fertilization (hpf) and describe early photoreceptor morphogenesis defects.

Genes / Markers

Figures

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Expression

Gene	Antibody	Fish	Conditions	Stage	Anatomy	Assay	Figure
ift52	ab1-ift52	ift172^{hi2211Tg/hi2211Tg}	standard conditions	Pec-fin	whole organism	WB	Fig. 6
ift52	ab1-ift52	ift172^{hi2211Tg/hi2211Tg}	standard conditions	Protruding-mouth	whole organism	WB	Fig. 8
ift52	ab1-ift52	ift57^{hi3417Tg/hi3417Tg}	standard conditions	Pec-fin	whole organism	WB	Fig. 6
ift52	ab1-ift52	ift57^{hi3417Tg/hi3417Tg}	standard conditions	Protruding-mouth	whole organism	WB	Fig. 8
ift52	ab1-ift52	ift88^tz288/tz288	standard conditions	Protruding-mouth	whole organism	WB	Fig. 8
ift52	ab1-ift52	ift88^tz288/tz288	standard conditions	Pec-fin	whole organism	WB	Fig. 6
ift52	ab1-ift52	WT	standard conditions	Pec-fin	whole organism	WB	Fig. 6
ift52	ab1-ift52	WT	standard conditions	Protruding-mouth	whole organism	WB	Fig. 8
ift88	ab1-ift88	ift172^{hi2211Tg/hi2211Tg}	standard conditions	Protruding-mouth	whole organism	WB	Fig. 8
ift88	ab1-ift88	ift172^{hi2211Tg/hi2211Tg}	standard conditions	Pec-fin	whole organism	WB	Fig. 6

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Phenotype

Fish	Conditions	Stage	Phenotype	Figure
ift172^{hi2211Tg/hi2211Tg}	standard conditions	Pec-fin	eye photoreceptor cell photoreceptor inner segment membrane disorganized, abnormal	Fig. 1
ift172^{hi2211Tg/hi2211Tg}	standard conditions	Pec-fin	eye photoreceptor cell photoreceptor outer segment absent, abnormal	Fig. 1
ift172^{hi2211Tg/hi2211Tg}	standard conditions	Pec-fin	retinal cone cell morphology, abnormal	Fig. 5
ift172^{hi2211Tg/hi2211Tg}	standard conditions	Protruding-mouth	eye photoreceptor cell centriole displaced, abnormal	Fig. 2
ift172^{hi2211Tg/hi2211Tg}	standard conditions	Protruding-mouth	eye photoreceptor cell photoreceptor connecting cilium absent, abnormal	Fig. 2
ift172^{hi2211Tg/hi2211Tg}	standard conditions	Protruding-mouth	eye photoreceptor cell photoreceptor inner segment membrane disorganized, abnormal	Fig. 2
ift172^{hi2211Tg/hi2211Tg}	standard conditions	Protruding-mouth	eye photoreceptor cell photoreceptor inner segment membrane displaced, abnormal	Fig. 2
ift172^{hi2211Tg/hi2211Tg}	standard conditions	Protruding-mouth	eye photoreceptor cell photoreceptor outer segment absent, abnormal	Fig. 2
ift172^{hi2211Tg/hi2211Tg}	standard conditions	Protruding-mouth	retinal cone cell morphology, abnormal	Fig. 7
ift172^{hi2211Tg/hi2211Tg}	standard conditions	Day 4	eye photoreceptor cell nucleus decreased length, abnormal	Fig. 3

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Mutations / Transgenics

Allele	Construct	Type	Affected Genomic Region
hi2211Tg	Tg(nLacz-GTvirus)	Transgenic Insertion	ift172
hi3417Tg	Tg(nLacz-GTvirus)	Transgenic Insertion	ift57
tz288		Point Mutation	ift88

Human Disease / Model

Sequence Targeting Reagents

Fish

Fish
ift57^{hi3417Tg/hi3417Tg}
ift88^tz288/tz288
ift172^{hi2211Tg/hi2211Tg}
WT

Antibodies

Orthology

Engineered Foreign Genes

Mapping

Sukumaran et al., 2009 ZDB-PUB-090116-17 PMID:19136023

Early defects in photoreceptor outer segment morphogenesis in zebrafish ift57, ift88 and ift172 Intraflagellar Transport mutants

Sukumaran et al., 2009

ZDB-PUB-090116-17
PMID:19136023