Language

English

Publication Date

5-23-2025

Journal

Science Advances

DOI

10.1126/sciadv.adv1281

PMID

40408471

PMCID

PMC12101511

PubMedCentral® Posted Date

5-23-2025

PubMedCentral® Full Text Version

Post-print

Abstract

Action potentials are initiated and modulated at the axon initial segment (AIS) by highly clustered ion channels. Voltage-gated Kv1 potassium channels underlie most outward AIS K+ current. AIS Kv1 channels exist in a large protein complex including ADAM22, Caspr2, and LGI1. However, their clustering mechanisms remain unknown. Because Kv1 channels have a highly conserved PDZ-binding motif, we used CRISPR-based genome editing to screen 18 PDZ domain-containing proteins identified in our previous AIS proximity proteome for their AIS localization. Among these, we found that the scaffolding proteins SCRIB and PSD93 are highly enriched at the AIS. Using CRISPR-mediated knockout, cell surface clustering assays, and coimmunoprecipitation, we show that SCRIB and PSD93 bind to and are required for AIS Kv1 channel clustering, whereas SCRIB links the AIS Kv1 channel protein complex to the master AIS scaffolding protein AnkyrinG. These results define a hierarchy of scaffolding proteins that combine to cluster AIS Kv1 channels.

Keywords

PDZ Domains, Animals, Shaker Superfamily of Potassium Channels, Protein Binding, Humans, Axon Initial Segment, Rats, Membrane Proteins, HEK293 Cells, Axons, Kv Channel-Interacting Proteins

Published Open-Access

yes

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