ZFIN ID: ZDB-FIG-170227-1
Lessieur et al., 2017 - The Ciliopathy Gene ahi1 Is Required for Zebrafish Cone Photoreceptor Outer Segment Morphogenesis and Survival. Investigative ophthalmology & visual science   58:448-460 Full text @ Invest. Ophthalmol. Vis. Sci.
Anatomical Terms:
Stage Range: 1-cell to Day 5
Observed In:
Stage: Day 5

Fig. 1

Generation of ahi1 mutant zebrafish. (A–C) Whole-mount in situ hybridization from 1 cell (A), 10 hpf (B), and 48 hpf (C), showing expression of ahi1. (D, E) Lateral and ventral views showing 5 dpf zebrafish stained by whole-mount in situ hybridization. ahi1 expression was observed throughout the central nervous system (arrowheads) and pronephros (arrow). (F) A transverse section shows ahi1 expression throughout the retina at 5 dpf. (G) Illustration of ahi1 genomic architecture. The wild-type sequence and TALENs induced ahi1 mutant alleles are shown. Wild-type nucleotides are in upper case. Deleted nucleotides are denoted as dashes while inserted nucleotides are in lower case letters. (H) Schematic protein structure of Ahi1 illustrating the location of the coiled-coil (CC) domain, WD40 repeats, and SH3 domain. The ahi1lri46 is a frame shift mutation and encodes a truncated protein lacking the 7 WD 40 repeats and the SH3 domain. (I) Chromatograms of Sanger sequencing reactions of ahi1 wild type (wild-type) and homozygous mutant (ahi1lri46) zebrafish. (J) Lateral view of representative wild-type and ahi1lri46 homozygous mutant at 5 dpf.

Gene Expression Details
Gene Antibody Fish Conditions Stage Anatomy Assay
ahi1 WT standard conditions 1-cell whole organism ISH
Bud whole organism ISH
Long-pec central nervous system ISH
Day 5 central nervous system ISH
Day 5 pronephros ISH
Day 5 retinal neural layer ISH
Antibody Labeling Details No data available
Phenotype Details
Fish Conditions Stage Phenotype
ahi1lri46/lri46 standard conditions Day 5 inner ear has normal numbers of parts of type otolith, normal
Day 5 post-vent region curved ventral, abnormal
Day 5 whole organism lacks all parts of type swim bladder, abnormal
ZFIN wishes to thank the journal Investigative ophthalmology & visual science for permission to reproduce figures from this article. Please note that this material may be protected by copyright. Full text @ Invest. Ophthalmol. Vis. Sci.