Functional studies of deafness-associated pendrin and prestin variants

S Takahashi, T Kojima, K Wasano… - International Journal of …, 2024 - mdpi.com
Pendrin and prestin are evolutionary-conserved membrane proteins that are essential for
normal hearing. Dysfunction of these proteins results in hearing loss in humans, and …

The pendrin polypeptide

S Dossena, E Bernardinelli, AK Sharma… - The Role of Pendrin in …, 2017 - Springer
Abstract Pendrin (SLC26A4) is an electroneutral anion exchanger in which function-altering
mutations cause inherited forms of non-syndromic (DFNB4) and syndromic deafness …

Reduction of cellular expression levels is a common feature of functionally affected pendrin (SLC26A4) protein variants

VCS de Moraes, E Bernardinelli, N Zocal… - Molecular …, 2016 - Springer
Sequence alterations in the pendrin gene (SLC26A4) leading to functionally affected protein
variants are frequently involved in the pathogenesis of syndromic and nonsyndromic …

The pathogenic roles of the p. R130S prestin variant in DFNB61 hearing loss

S Takahashi, Y Zhou, MA Cheatham… - The Journal of …, 2024 - Wiley Online Library
DFNB61 is a recessively inherited nonsyndromic hearing loss caused by mutations in
SLC26A5, the gene that encodes the voltage‐driven motor protein, prestin. Prestin is …

Prestin, a cochlear motor protein, is defective in non-syndromic hearing loss

XZ Liu, XM Ouyang, XJ Xia, J Zheng… - Human molecular …, 2003 - academic.oup.com
Prestin, a membrane protein that is highly and almost exclusively expressed in the outer hair
cells (OHCs) of the cochlea, is a motor protein which senses membrane potential and drives …

High frequency of the IVS2-2A>G DNA sequence variation in SLC26A5, encoding the cochlear motor protein prestin, precludes its involvement in hereditary hearing …

HY Tang, A Xia, JS Oghalai, FA Pereira… - BMC Medical Genetics, 2005 - Springer
Background Cochlear outer hair cells change their length in response to variations in
membrane potential. This capability, called electromotility, is believed to enable the …

The roles of conserved and nonconserved cysteinyl residues in the oligomerization and function of mammalian prestin

B Currall, D Rossino… - Journal of …, 2011 - journals.physiology.org
The creation of several prestin knockout and knockin mouse lines has demonstrated the
importance of the intrinsic outer hair cell membrane protein prestin to mammalian hearing …

Essential helix interactions in the anion transporter domain of prestin revealed by evolutionary trace analysis

L Rajagopalan, N Patel, S Madabushi… - Journal of …, 2006 - Soc Neuroscience
Prestin, a member of the SLC26A family of anion transporters, is a polytopic membrane
protein found in outer hair cells (OHCs) of the mammalian cochlea. Prestin is an essential …

The R130S mutation significantly affects the function of prestin, the outer hair cell motor protein

S Takahashi, MA Cheatham, J Zheng… - Journal of Molecular …, 2016 - Springer
Abstract A missense mutation, R130S, was recently found in the prestin gene, SLC26A5, of
patients with moderate to severe hearing loss (DFNB61). In order to define the pathology of …

Engineered pendrin protein, an anion transporter and molecular motor

J Tang, JL Pecka, X Tan, KW Beisel, DZZ He - Journal of Biological …, 2011 - ASBMB
Pendrin and prestin both belong to a distinct anion transporter family called solute carrier
protein 26A, or SLC26A. Pendrin (SLC26A4) is a chloride-iodide transporter that is found at …