ZFIN ID: ZDB-FIG-121206-2
Obholzer et al., 2012 - Rapid positional cloning of zebrafish mutations by linkage and homozygosity mapping using whole-genome sequencing. Development (Cambridge, England)   139(22):4280-4290 Full text @ Development
ADDITIONAL FIGURES
EXPRESSION / LABELING:
Genes:
Fish:
Anatomical Terms:
Stage: Pec-fin
PHENOTYPE:
Fish:
Knockdown Reagents:
Observed In:
Stage: Pec-fin

Fig. 4 Validation of the identified ear mutants. (A) Lateral view of a wild-type ear at 65 hpf (fused projections labeled). Ant., anterior projection; Lat., lateral projection; Pst., posterior projection; Vnt., ventral projection. (B) Semicircular canal projections are abnormally shaped in jj410 ears at 65 hpf. (C) lmx1b morpholino knockdown recapitulates jj410 ear phenotype at 65 hpf. (D) lmx1b antisense in situ probe marks the inner ear and concentrates in semicircular canal projections. (E) Severe reduction of lmx1b transcript levels in a jj410 mutant larval inner ear at 65 hpf. (F) Sequencing trace of the jj410 genomic lesion in lmx1b; arrow denotes causative mutation that introduces a premature stop codon TGA (underlined, antisense strand was sequenced). (G) Control morpholino injection recapitulates wild type. (H) jj59 inner ears are shorter in the anterior-posterior axis and semicircular canal projections are malformed at 65 hpf. (I) jag1b morpholino knockdown recapitulates jj59 ear phenotype at 65 hpf. (J) jag1b antisense in situ probe marks the inner ear and concentrates in semicircular canal projections. (K) Severe reduction of jag1b transcript levels in a jj59 mutant larval inner ear at 65 hpf. (L) Sequencing trace of the jj59 genomic lesion in jag1b; arrow denotes causative mutation. (M) YO-PRO1 uptake by neuromast hair cells in wild-type larvae. (N) an158-3(cdh23nl9) mutant larval neuromast hair cells fail to take up YO-PRO1. (O) Failure to complement YO-PRO1 uptake in an158-3 x tc317e larval hair cells. (P) Intact inner ear hair cell bundles in the wild type. (Q) Hair cell bundles are splayed in an158-3(cdh23nl9) mutant inner ear hair cells. (R) Hair cell bundles show splayed phenotype and thus fails to complement in an158-3 x tc317e mutant inner ear. (S) Sequencing trace of the an158-3/cdh23nl9 genomic lesion in cdh23 (arrow).

Gene Expression Details
Gene Antibody Fish Conditions Stage Qualifier Anatomy Assay
jag1b AB standard conditions Pec-fin inner ear ISH
Pec-fin otic vesicle protrusion ISH
jag1bjj59/jj59 (AB) standard conditions Pec-fin Not Detected inner ear ISH
lmx1bb AB standard conditions Pec-fin inner ear ISH
Pec-fin otic vesicle protrusion ISH
lmx1bbjj410/jj410 (AB) standard conditions Pec-fin inner ear ISH
Antibody Labeling Details No data available
Phenotype Details
Fish Conditions Stage Phenotype
AB + MO2-lmx1bb standard conditions Pec-fin otic vesicle protrusion malformed, abnormal
Pec-fin otic vesicle protrusion shape, abnormal
AB + MO6-jag1b standard conditions Pec-fin otic vesicle anterior-posterior axis decreased length, abnormal
Pec-fin otic vesicle protrusion malformed, abnormal
Pec-fin otic vesicle protrusion shape, abnormal
cdh23nl9/+; cdh23tc317e/+ standard conditions Pec-fin auditory receptor cell stereocilium bundle splayed, abnormal
Pec-fin neuromast hair cell morphology, abnormal
cdh23nl9/nl9 standard conditions Pec-fin auditory receptor cell stereocilium bundle splayed, abnormal
Pec-fin neuromast hair cell morphology, abnormal
jag1bjj59/jj59 (AB) standard conditions Pec-fin otic vesicle anterior-posterior axis decreased length, abnormal
Pec-fin otic vesicle protrusion malformed, abnormal
Pec-fin otic vesicle protrusion shape, abnormal
lmx1bbjj410/jj410 (AB) standard conditions Pec-fin otic vesicle protrusion malformed, abnormal
Pec-fin otic vesicle protrusion shape, abnormal
Acknowledgments:
This image is the copyrighted work of the attributed author or publisher, and ZFIN has permission only to display this image to its users. Additional permissions should be obtained from the applicable author or publisher of the image. Full text @ Development