ZFIN ID: ZDB-FIG-140530-3
Blanco et al., 2014 - Complexes of Usher proteins preassemble at the endoplasmic reticulum and are required for trafficking and ER homeostasis. Disease models & mechanisms   7:547-59 Full text @ Dis. Model. Mech.
ADDITIONAL FIGURES
EXPRESSION / LABELING:
Genes: ift88, ush1c
Antibodies: Ab1-cdh23, Ab1-ift88, Ab2-myo7a
Fish: WT
Anatomical Terms: hair cell, hair cell kinocilium
Stage: Day 5
PHENOTYPE:
Fish: cdh23tj264/tj264, ift88tz288/tz288, myo7aaty220/ty220, ush1cfh293/fh293
Observed In: anterior macula stereocilium bundle, hair cell anterior macula, hair cell anterior macula apoptotic process, hair cell anterior macula epithelium, hair cell anterior macula stereocilium
Stage: Day 5

Fig. 1 Zebrafish cdh23, ush1c, myo7aa and ift88 mutants have mechanoreceptor structural defects. (A–E) Confocal projections of anterior maculae labeled with phalloidin in (A) wild-type (WT) sibling, (B) cdh23tj264a, (C) ush1cfh293, (D) ift88tz288b and (E) myo7aaty220d mutants. (F–H) Confocal optical sections of double immunolabeling of (F) Harmonin (green) and Ift88 (red), (G) Harmonin (green) and Cdh23 (red), and (H) Harmonin (green) and Myo7aa (red). Individual hair cells are shown. (I) Quantification of total number of hair bundles. ne6 analyzed anterior maculae. (J) Quantification of total number of hair cells. ne5 analyzed anterior maculae. (K) Quantification of hair cell death in mutants and wild-type siblings. Number: average number of TUNEL-positive hair cells per macula. ne16 analyzed anterior maculae. Student’s t-test: **P<0.01, *P<0.05. NS, non significant. Black lines represent ± s.e.m. Scale bars: 5 µm.

Gene Expression Details
Gene Antibody Fish Conditions Stage Anatomy Assay
ift88 Ab1-ift88 WT standard conditions Day 5 hair cell IHC
Ab1-ift88 Day 5 hair cell kinocilium IHC
ush1c WT standard conditions Day 5 hair cell IHC
Day 5 hair cell kinocilium IHC
Antibody Labeling Details
Antibody Assay Fish Conditions Stage Anatomy
Ab1-cdh23 IHC WT standard conditions Day 5 hair cell
Ab2-myo7a IHC WT standard conditions Day 5 hair cell
Phenotype Details
Fish Conditions Stage Phenotype
cdh23tj264/tj264 standard conditions Day 5 anterior macula stereocilium bundle bent, abnormal
Day 5 anterior macula stereocilium bundle splayed, abnormal
Day 5 hair cell anterior macula normal amount, normal
Day 5 hair cell anterior macula apoptotic process increased occurrence, abnormal
Day 5 hair cell anterior macula epithelium organization quality, normal
Day 5 hair cell anterior macula stereocilium decreased amount, abnormal
ift88tz288/tz288 standard conditions Day 5 anterior macula stereocilium bundle bent, abnormal
Day 5 anterior macula stereocilium bundle splayed, abnormal
Day 5 hair cell anterior macula normal amount, normal
Day 5 hair cell anterior macula apoptotic process increased occurrence, abnormal
Day 5 hair cell anterior macula epithelium organization quality, normal
Day 5 hair cell anterior macula stereocilium decreased amount, abnormal
myo7aaty220/ty220 standard conditions Day 5 anterior macula stereocilium bundle bent, abnormal
Day 5 anterior macula stereocilium bundle splayed, abnormal
Day 5 hair cell anterior macula stereocilium decreased amount, abnormal
ush1cfh293/fh293 standard conditions Day 5 anterior macula stereocilium bundle bent, abnormal
Day 5 anterior macula stereocilium bundle splayed, abnormal
Day 5 hair cell anterior macula stereocilium 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.