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Structures of the junctophilin/voltage-gated calcium channel interface reveal hot spot for cardiomyopathy mutations

Zheng Fang Yang, Pankaj Panwar, Ciaran McFarlane, Wietske E. Tuinte, Marta Campiglio, Filip Van Petegem

2022Proceedings of the National Academy of Sciences35 citationsDOIOpen Access PDF

Abstract

SignificanceIon channels have evolved the ability to communicate with one another, either through protein-protein interactions, or indirectly via intermediate diffusible messenger molecules. In special cases, the channels are part of different membranes. In muscle tissue, the T-tubule membrane is in proximity to the sarcoplasmic reticulum, allowing communication between L-type calcium channels and ryanodine receptors. This process is critical for excitation-contraction coupling and requires auxiliary proteins like junctophilin (JPH). JPHs are targets for disease-associated mutations, most notably hypertrophic cardiomyopathy mutations in the JPH2 isoform. Here we provide high-resolution snapshots of JPH, both alone and in complex with a calcium channel peptide, and show how this interaction is targeted by cardiomyopathy mutations.

Topics & Concepts

Ryanodine receptorEndoplasmic reticulumVoltage-dependent calcium channelCalcium signalingCalcium channelBiologyBiophysicsIon channelCalciumCell biologyGene isoformCardiac muscleInternal medicineChemistryReceptorEndocrinologyGeneticsGeneSignal transductionMedicineIon channel regulation and functionCardiomyopathy and Myosin StudiesCardiac electrophysiology and arrhythmias