Transcription factor Emx2 controls stereociliary bundle orientation of sensory hair cells

Jiang, Tao; Kindt, Katie S.; Wu, Doris K.

doi:10.7554/elife.23661

Cited by 113 publications

(265 citation statements)

References 76 publications

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“…2). Recent reports show that Emx2 and Notch signaling control stereocolia bundle orientation and that Vangl2 is normally localized in emx2 mutants suggesting that this pathway acts in parallel to the PCP pathway (Jacobo A, 2018;Jiang et al, 2017). Emx2 is expressed in only one of the pair of sister hair cells and changes its polarity by 180 degrees, thereby causing the pair of hair cells to adopt opposing polarities.…”

Section: Pcp and Wnt Signaling Pathways Control Two Different Aspectsmentioning

confidence: 99%

“…1c). The positioning of the kinocilium along this axis is controlled by Notch signaling/Emx2 and its loss causes all hair cells within a neuromast to point into the same direction (Jacobo A, 2018;Jiang et al, 2017). Hair cells within sensory organs show different polarities of this common axis depending on which placode/primordium they originate from, allowing the animal to sense water motion coming from all directions ( Fig.…”

Section: Introductionmentioning

confidence: 99%

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Parallel control of mechanosensory hair cell orientation by the PCP and Wnt pathways

Acedo

Voas

Richard

et al. 2019

Preprint

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Cell polarity plays a crucial role during development of vertebrates and invertebrates.Planar Cell Polarity (PCP) is defined as the coordinated polarity of cells within a tissue axis and is essential for processes such as gastrulation, neural tube closure or hearing. Wnt ligands can be instructive or permissive during PCP-dependent processes, and Wnt pathway mutants are often classified as PCP mutants due to the complexity and the similarities between their phenotypes.Our studies of the zebrafish sensory lateral line reveal that disruptions of the PCP and Wnt pathways have differential effects on hair cell orientations. While mutations in PCP genes cause random orientations of hair cells, mutations in Wnt pathway members induce hair cells to adopt a concentric pattern. We show that PCP signaling is normal in hair cells of Wnt pathway mutants and that the concentric hair cell phenotype is due to altered organization of the surrounding support cells. Thus, the PCP and Wnt pathways work in parallel, as separate pathways to establish proper hair cell orientation. Our data suggest that coordinated support cell organization is established during the formation of lateral line primordia, much earlier than the appearance of hair cells. Together, these finding reveal that hair cell orientation defects are not solely explained by defects in PCP signaling and that some hair cell phenotypes warrant reevaluation.

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Section: Pcp and Wnt Signaling Pathways Control Two Different Aspectsmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Parallel control of mechanosensory hair cell orientation by the PCP and Wnt pathways

Acedo

Voas

Richard

et al. 2019

Preprint

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“…1a, Supplemental Fig. 5) 34 . We determined that the relative position of the LPR was not altered in either the mutant utricle or saccule (Supplemental Fig.…”

Section: Anatomical Features Of the Striola Are Affected By Cyp26b1 Cmentioning

confidence: 99%

Cytochrome P450 26b1-mediated specification of vestibular striola and central zones is required for transient responses in linear acceleration

Ono

Keller

Ramírez

et al. 2019

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Each vestibular sensory epithelia of the inner ear is divided into two zones, the striola and extrastriola in maculae of otolith organs and the central and peripheral zones in cristae of semicircular canals, that differ in morphology and physiology. We found that formation of striolar/central zones during embryogenesis requires Cytochrome P450 26b1 (Cyp26b1)-mediated degradation of retinoic acid (RA). In Cyp26b1 conditional knockout mice, the identities of the striolar/central zones were compromised, including abnormal innervating neurons and otoconia in otolith organs. Vestibular evoked potentials (VsEP) in response to jerk stimuli were largely absent. Vestibulo-ocular reflexes and standard motor performances such as forced swimming were unaffected, but mutants had head tremors and deficits in balance beam tests that were consistent with abnormal vestibular input. Thus, degradation of RA during embryogenesis is required for patterning highly specialized regions of the vestibular sensory epithelia that may provide acute feedback about head motion.

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“…Ca 2+ signals at the base of hair cells(Jiang et al, 2017a;Sheets et al, 2017), and concurrently 190 used MitoRGECO1 (red) to examine mito-Ca 2+ signals(Figure 2A-B'). Our two-color imaging 191 approach revealed a strong correlation between the magnitude of the GCaMP6sCAAX and 192MitoRGECO1 signals (Figure 2B, R 2 = 0.8, p < 0.0001; n = 209 cells).…”

mentioning

confidence: 99%

Synaptic mitochondria are critical for hair-cell synapse formation and function

Wong

Zhang

Beirl

et al. 2019

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Sensory hair cells in the ear utilize specialized ribbon synapses. These synapses are 22 defined by electron-dense presynaptic structures called ribbons, composed primarily of the 23 structural protein Ribeye. Previous work has shown that voltage-gated influx of Ca 2+ through 24CaV1.3 channels is critical for hair-cell synapse function and can impede ribbon formation. We 25show that in mature zebrafish hair cells, evoked presynaptic-Ca 2+ influx through CaV1.3 channels 26 initiates mitochondrial-Ca 2+ (mito-Ca 2+ ) uptake adjacent to ribbons. Block of mito-Ca 2+ uptake in 27 mature cells depresses presynaptic Ca 2+ influx and impacts synapse integrity. In developing 28 zebrafish hair cells, mito-Ca 2+ uptake coincides with spontaneous rises in presynaptic Ca 2+ influx. 29Spontaneous mito-Ca 2+ loading lowers cellular NAD + /NADH redox and downregulates ribbon 30 formation. Direct application of NAD + or NADH increases or decreases ribbon formation 31 respectively, possibly acting through the NAD(H)-binding domain on Ribeye. Our results present 32 a mechanism where presynaptic-and mito-Ca 2+ couple to confer proper presynaptic function 33 and formation. 34 35

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Transcription factor Emx2 controls stereociliary bundle orientation of sensory hair cells

Cited by 113 publications

References 76 publications

Parallel control of mechanosensory hair cell orientation by the PCP and Wnt pathways

Parallel control of mechanosensory hair cell orientation by the PCP and Wnt pathways

Cytochrome P450 26b1-mediated specification of vestibular striola and central zones is required for transient responses in linear acceleration

Synaptic mitochondria are critical for hair-cell synapse formation and function

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