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Excessive EFHD1-dependent ER-mitochondrial contacts drive a maladaptive antiviral response in metabolic liver disease

David R. Eberhardt, Emma C. Rekate, Yasmin B. Masini, Hannah E. Duron, David Mollinedo, Adrian M. Velarde, Devorah Stucki, Tara R. Price, Sandra H.J. Lee, Enrique Balderas, Neeraj K. Rai, Ashley R. Bratt, Anthony M. Balynas, Chris J. Stubben, Ryan Bia, Sudipa Maity, Nicolas Hartel, Xue Yin, Andrea Corbin, Anshu Kumari, Dung M. Nguyen, Daisuke Shimura, Vu D. Nguyen, Vishaka Vinod, Kamrul H. Chowdhury, Francisco Verdeguer, Joel Zvick, Patrice N. Mimche, Sihem Boudina, Stavros G. Drakos, Ademuyiwa S. Aromolaran, Sarah Franklin, Vivek Garg, Robin M. Shaw, William L. Holland, Scott A. Summers, Marcus G. Pezzolesi, Jared Rutter, Kimberley J. Evason, Dipayan Chaudhuri

Journal of Clinical Investigation · 2026

Vollständiger Abstract

Worum geht es in dieser Arbeit?

Metabolic-associated steatohepatitis (MASH) involves hepatocyte damage that cannot be explained solely by lipid accumulation. Here, to discover injury-specific pathways, we focused on a gene of uncertain function, EF-Hand Domain Family Member D1 (EFHD1), identified in human genome-wide association studies of liver injury but not liver fat. We show that EFHD1, a Ca 2+ -dependent actin crosslinker, stabilizes endoplasmic reticulum–mitochondria contact sites (ERMCS), detecting spatiotemporal coincidence of inter-organellar proximity and ER Ca 2+ release. During MASH, EFHD1 upregulation drives pathological mitochondrial fragmentation via excessive contact persistence. This structural failure promotes mitochondrial double-stranded RNA escape and activation of a maladaptive antiviral PKR-associated stress response, a causal relationship also supported by Mendelian randomization in humans. Consequently, inhibiting EFHD1 in human and mouse models blunts hepatocyte damage. These findings identify EFHD1 as a Ca 2+ -dependent ERMCS stabilizer, reveal a hepatocyte-intrinsic injury pathway, and suggest EFHD1 inhibition as a therapeutic strategy.

Bibliografischer Nachweis

Publikationsdaten

Autor:innen
David R. Eberhardt, Emma C. Rekate, Yasmin B. Masini, Hannah E. Duron, David Mollinedo, Adrian M. Velarde, Devorah Stucki, Tara R. Price, Sandra H.J. Lee, Enrique Balderas, Neeraj K. Rai, Ashley R. Bratt, Anthony M. Balynas, Chris J. Stubben, Ryan Bia, Sudipa Maity, Nicolas Hartel, Xue Yin, Andrea Corbin, Anshu Kumari, Dung M. Nguyen, Daisuke Shimura, Vu D. Nguyen, Vishaka Vinod, Kamrul H. Chowdhury, Francisco Verdeguer, Joel Zvick, Patrice N. Mimche, Sihem Boudina, Stavros G. Drakos, Ademuyiwa S. Aromolaran, Sarah Franklin, Vivek Garg, Robin M. Shaw, William L. Holland, Scott A. Summers, Marcus G. Pezzolesi, Jared Rutter, Kimberley J. Evason, Dipayan Chaudhuri
Quelle
Journal of Clinical Investigation
Publikation
2026-01-01
Band / Ausgabe
Nicht angegeben
Seiten
Nicht angegeben
ISSN / ISBN
1558-8238
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Zitierfähiger Nachweis

David R. Eberhardt, Emma C. Rekate, Yasmin B. Masini, Hannah E. Duron, David Mollinedo, Adrian M. Velarde, Devorah Stucki, Tara R. Price, Sandra H.J. Lee, Enrique Balderas, Neeraj K. Rai, Ashley R. Bratt, Anthony M. Balynas, Chris J. Stubben, Ryan Bia, Sudipa Maity, Nicolas Hartel, Xue Yin, Andrea Corbin, Anshu Kumari, Dung M. Nguyen, Daisuke Shimura, Vu D. Nguyen, Vishaka Vinod, Kamrul H. Chowdhury, Francisco Verdeguer, Joel Zvick, Patrice N. Mimche, Sihem Boudina, Stavros G. Drakos, Ademuyiwa S. Aromolaran, Sarah Franklin, Vivek Garg, Robin M. Shaw, William L. Holland, Scott A. Summers, Marcus G. Pezzolesi, Jared Rutter, Kimberley J. Evason, Dipayan Chaudhuri (2026). Excessive EFHD1-dependent ER-mitochondrial contacts drive a maladaptive antiviral response in metabolic liver disease. Journal of Clinical Investigation. https://doi.org/10.1172/jci204023
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