ZFIN ID: ZDB-FIG-111215-36
Phillips et al., 2011 - Harmonin (Ush1c) is required in zebrafish Muller glial cells for photoreceptor synaptic development and function. Disease models & mechanisms   4(6):786-800 Full text @ Dis. Model. Mech.
ADDITIONAL FIGURES
EXPRESSION / LABELING:
Gene: ush1c
Antibody: Ab1-ush1c
Fish: AB, ush1cfh293/fh293, AB + MO1-ush1c
Knockdown Reagent: MO1-ush1c
Anatomical Terms: anterior crista, anterior macula, lateral crista, neuromast, otic sensory epithelium (all 7) expand
Stage: Protruding-mouth
PHENOTYPE:
Fish: ush1cfh293/fh293, WT + MO1-ush1c
Knockdown Reagent: MO1-ush1c
Observed In: auditory receptor cell kinocilium, auditory receptor cell stereocilium bundle, hair cell anterior macula, hair cell posterior macula
Stage: Day 5

Fig. 3 Harmonin is required in otic sensory patches for normal bundle development. (A–C) Phalloidin-stained F-actin (green) and anti-zebrafish harmonin antibody labeling (red) in 80-hpf wild-type ears show localization of harmonin relative to the stereocilia in the five otic sensory patches (ac, anterior crista; am, anterior macula; lc, lateral crista; pc, posterior crista; pm, posterior macula) and in the neuromasts (n). The anti-harmonin antibody signal is reduced in sensory patches and in neuromasts in ush1c mutants (B) and ush1c-MO-injected larvae (C). (D–L) High-magnification view of phalloidin-stained hair bundles in the anterior and posterior maculae of 5 dpf larvae. Hair bundle number is reduced and organization of the existing bundles is disrupted in mutant (G–I) and morphant (J–L) anterior (G,J) and posterior (H,K) maculae, compared with the anterior and posterior maculae of wild-type controls (D and E, respectively). Closeup images of hair bundles of the anterior maculae (F,I,L) show bent and splayed bundles in mutants and morphants. For mutant analysis, anterior macular wild-type average=54.6±3.5 bundles; n=8; ush1c mutant average=33±3.5; n=7; P<0.0001. Posterior macula wild-type average=68.2±2.6 bundles; n=9; ush1c mutant average=40.5±4.2; n=10; P<0.0001. For the morpholino experiment, anterior macula control average=41.8±4.8 bundles; MO average=21.8±3.8; n=5 for each group; P<0.0001. Posterior macula control average=71.8±4.6 bundles; MO average=48.2±6.9; n=5 for each; P<0.0001, 5 dpf. Scale bars: 50 μm (A–C); 10 μm (D–L).

Gene Expression Details
Gene Antibody Fish Conditions Stage Anatomy Assay
ush1c Ab1-ush1c AB standard conditions Protruding-mouth anterior crista IHC
Ab1-ush1c Protruding-mouth anterior macula IHC
Ab1-ush1c Protruding-mouth lateral crista IHC
Ab1-ush1c Protruding-mouth neuromast IHC
Ab1-ush1c Protruding-mouth posterior crista IHC
Ab1-ush1c Protruding-mouth posterior macula IHC
Ab1-ush1c AB + MO1-ush1c standard conditions Protruding-mouth otic sensory epithelium IHC
Ab1-ush1c ush1cfh293/fh293 standard conditions Protruding-mouth otic sensory epithelium IHC
Antibody Labeling Details No data available
Phenotype Details
Fish Conditions Stage Phenotype
WT + MO1-ush1c standard conditions Day 5 auditory receptor cell kinocilium bent, abnormal
Day 5 auditory receptor cell stereocilium bundle splayed, abnormal
Day 5 hair cell anterior macula decreased amount, abnormal
Day 5 hair cell posterior macula decreased amount, abnormal
ush1cfh293/fh293 standard conditions Day 5 auditory receptor cell kinocilium bent, abnormal
Day 5 auditory receptor cell stereocilium bundle splayed, abnormal
Day 5 hair cell anterior macula decreased amount, abnormal
Day 5 hair cell posterior macula decreased amount, abnormal
Acknowledgments:
ZFIN wishes to thank the journal Disease models & mechanisms for permission to reproduce figures from this article. Please note that this material may be protected by copyright. Full text @ Dis. Model. Mech.