Wnt/PKC Signaling Inhibits Sensory Hair Cell Formation in the Developing Mammalian Cochlea

The establishment of cell fate and boundaries between cell types is an essential step in development and organogenesis. In the mammalian cochlea, a distinct boundary exists between a medial region of non-sensory cells and a lateral region of sensory cells. We report that Wnt4 and sFRP2 act in combin...

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Main Authors: Joanna F. Mulvaney, Erynn M. Layman, Farhana Feroze-Merzoug, Julia M. Abitbol, Jennifer M. Jones, Dara O’Connor, Florence Naillat, Seppo Vainio, Jeffrey S. Rubin, Matthew W. Kelley, Alain Dabdoub
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Language:English
Published: MDPI AG 2025-06-01
Series:Cells
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Online Access:https://www.mdpi.com/2073-4409/14/12/888
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author Joanna F. Mulvaney
Erynn M. Layman
Farhana Feroze-Merzoug
Julia M. Abitbol
Jennifer M. Jones
Dara O’Connor
Florence Naillat
Seppo Vainio
Jeffrey S. Rubin
Matthew W. Kelley
Alain Dabdoub
author_facet Joanna F. Mulvaney
Erynn M. Layman
Farhana Feroze-Merzoug
Julia M. Abitbol
Jennifer M. Jones
Dara O’Connor
Florence Naillat
Seppo Vainio
Jeffrey S. Rubin
Matthew W. Kelley
Alain Dabdoub
author_sort Joanna F. Mulvaney
collection DOAJ
description The establishment of cell fate and boundaries between cell types is an essential step in development and organogenesis. In the mammalian cochlea, a distinct boundary exists between a medial region of non-sensory cells and a lateral region of sensory cells. We report that Wnt4 and sFRP2 act in combination to modulate the sensory cell differentiation of the organ of Corti. The hair cell inhibitory effects of Wnt4 in the inner ear are mediated through the activation of the non-canonical Wnt/Calcium/PKC pathway. We show that Wnt4 stimulates the activation of PKC in the cochlea, and that the inhibition of PKC rescues the ectopic Wnt4 activity phenotype. Finally, we demonstrate that modification at a PKC target site on Atoh1 diminishes its ability to induce hair cell formation. Ultimately, we identify a new Wnt/Calcium/PKC non-canonical signaling pathway that is involved in proper hair cell and organ of Corti formation in the developing mammalian cochlea.
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series Cells
spelling doaj-art-bacea1f2ffcf41f2af563e62c4e1fd072025-08-20T03:27:09ZengMDPI AGCells2073-44092025-06-01141288810.3390/cells14120888Wnt/PKC Signaling Inhibits Sensory Hair Cell Formation in the Developing Mammalian CochleaJoanna F. Mulvaney0Erynn M. Layman1Farhana Feroze-Merzoug2Julia M. Abitbol3Jennifer M. Jones4Dara O’Connor5Florence Naillat6Seppo Vainio7Jeffrey S. Rubin8Matthew W. Kelley9Alain Dabdoub10Sunnybrook Research Institute, Sunnybrook Health Sciences Centre, 2075 Bayview Ave, Toronto, ON M4N 3M5, CanadaLaboratory of Cochlear Development, NIDCD, National Institutes of Health, Bethesda, MD 20892, USALaboratory of Cellular and Molecular Biology, NCI, National Institutes of Health, Bethesda, MD 20892, USASunnybrook Research Institute, Sunnybrook Health Sciences Centre, 2075 Bayview Ave, Toronto, ON M4N 3M5, CanadaDepartment of Biology, Saint Mary’s College, Notre Dame, IN 46556, USASunnybrook Research Institute, Sunnybrook Health Sciences Centre, 2075 Bayview Ave, Toronto, ON M4N 3M5, CanadaDepartment of Biochemistry and Molecular Medicine, Biocenter and Infotech Oulu, University of Oulu, FIN-90014 Oulu, FinlandDepartment of Biochemistry and Molecular Medicine, Biocenter and Infotech Oulu, University of Oulu, FIN-90014 Oulu, FinlandLaboratory of Cellular and Molecular Biology, NCI, National Institutes of Health, Bethesda, MD 20892, USALaboratory of Cochlear Development, NIDCD, National Institutes of Health, Bethesda, MD 20892, USASunnybrook Research Institute, Sunnybrook Health Sciences Centre, 2075 Bayview Ave, Toronto, ON M4N 3M5, CanadaThe establishment of cell fate and boundaries between cell types is an essential step in development and organogenesis. In the mammalian cochlea, a distinct boundary exists between a medial region of non-sensory cells and a lateral region of sensory cells. We report that Wnt4 and sFRP2 act in combination to modulate the sensory cell differentiation of the organ of Corti. The hair cell inhibitory effects of Wnt4 in the inner ear are mediated through the activation of the non-canonical Wnt/Calcium/PKC pathway. We show that Wnt4 stimulates the activation of PKC in the cochlea, and that the inhibition of PKC rescues the ectopic Wnt4 activity phenotype. Finally, we demonstrate that modification at a PKC target site on Atoh1 diminishes its ability to induce hair cell formation. Ultimately, we identify a new Wnt/Calcium/PKC non-canonical signaling pathway that is involved in proper hair cell and organ of Corti formation in the developing mammalian cochlea.https://www.mdpi.com/2073-4409/14/12/888mousehearingcochleageneshair cell
spellingShingle Joanna F. Mulvaney
Erynn M. Layman
Farhana Feroze-Merzoug
Julia M. Abitbol
Jennifer M. Jones
Dara O’Connor
Florence Naillat
Seppo Vainio
Jeffrey S. Rubin
Matthew W. Kelley
Alain Dabdoub
Wnt/PKC Signaling Inhibits Sensory Hair Cell Formation in the Developing Mammalian Cochlea
Cells
mouse
hearing
cochlea
genes
hair cell
title Wnt/PKC Signaling Inhibits Sensory Hair Cell Formation in the Developing Mammalian Cochlea
title_full Wnt/PKC Signaling Inhibits Sensory Hair Cell Formation in the Developing Mammalian Cochlea
title_fullStr Wnt/PKC Signaling Inhibits Sensory Hair Cell Formation in the Developing Mammalian Cochlea
title_full_unstemmed Wnt/PKC Signaling Inhibits Sensory Hair Cell Formation in the Developing Mammalian Cochlea
title_short Wnt/PKC Signaling Inhibits Sensory Hair Cell Formation in the Developing Mammalian Cochlea
title_sort wnt pkc signaling inhibits sensory hair cell formation in the developing mammalian cochlea
topic mouse
hearing
cochlea
genes
hair cell
url https://www.mdpi.com/2073-4409/14/12/888
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