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Keywords: Myo7aa
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Journal Articles

L-type voltage-gated calcium channel agonists mitigate hearing loss and modify ribbon synapse morphology in the zebrafish model of Usher syndrome type 1

In collection:
Developmental Disorders , Rare Disease Translational Research , Zebrafish as a Disease Model
Alaa Koleilat, Joseph A. Dugdale, Trace A. Christenson, Jeffrey L. Bellah, Aaron M. Lambert, Mark A. Masino, Stephen C. Ekker, Lisa A. Schimmenti
Journal: Disease Models & Mechanisms
Dis Model Mech (2020) 13 (11): dmm043885.
https://doi.org/10.1242/dmm.043885
Published: 27 November 2020
...Alaa Koleilat; Joseph A. Dugdale; Trace A. Christenson; Jeffrey L. Bellah; Aaron M. Lambert; Mark A. Masino; Stephen C. Ekker; Lisa A. Schimmenti ABSTRACT The mariner ( myo7aa −/− ) mutant is a zebrafish model for Usher syndrome type 1 (USH1). To further characterize hair cell synaptic elements...
View article titled, L-type voltage-gated calcium channel agonists mitigate hearing loss and modify ribbon synapse morphology in the zebrafish model of Usher syndrome type 1
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Includes: Supplementary data
Journal Articles

Complexes of Usher proteins preassemble at the endoplasmic reticulum and are required for trafficking and ER homeostasis

Bernardo Blanco-Sánchez, Aurélie Clément, Javier Fierro, Jr, Philip Washbourne, Monte Westerfield
Journal: Disease Models & Mechanisms
Dis Model Mech (2014) 7 (5): 547–559.
https://doi.org/10.1242/dmm.014068
Published: 1 May 2014
... genes produce USH, but the proximal cause of sensory cell death remains mysterious. We adapted a proximity ligation assay to analyze associations among three of the USH proteins, Cdh23, Harmonin and Myo7aa, and the microtubule-based transporter Ift88 in zebrafish inner ear mechanosensory hair cells. We...
View article titled, Complexes of Usher proteins preassemble at the endoplasmic reticulum and are required for trafficking and ER homeostasis
Open the PDF for Complexes of Usher proteins preassemble at the endoplasmic reticulum and are required for trafficking and ER homeostasis in another window
Includes: Supplementary data