Cystathionine γ-lyase is a major regulator of cognitive function through neurotrophin signaling and neurogenesis

S Suwarna Chakraborty (Department of Physiology, Pharmacology, & Therapeutics, Johns Hopkins University School of Medicine) S Sunil Jamuna Tripathi (Department of Physiology, Pharmacology, & Therapeutics, Johns Hopkins University School of Medicine) E Edwin Vázquez-Rosa (Department of Psychiatry, Case Western Reserve University) K Kalyani Chaubey (Department of Psychiatry, Case Western Reserve University) H Hisashi Fujioka (Cryo-Electron Microscopy Core Facility, Case Western Reserve University School of Medicine) E Emiko Miller (Department of Psychiatry, Case Western Reserve University) R Richa Tyagi (The Solomon H. Snyder Department of Neuroscience, Johns Hopkins University School of Medicine) T Thibaut Vignane (Leibniz Institute for Analytical Sciences) S Sudarshana M. Sharma (Department of Biochemistry and Molecular Biology and Hollings Cancer Center, Medical University of South Carolina) B Bobby Thomas (Darby Children’s Research Institute, Medical University of South Carolina) Z Zachary M. Weil (Department of Neuroscience, Rockefeller Neuroscience Institute, West Virginia University School of Medicine) R Randy J. Nelson (Department of Neuroscience, Rockefeller Neuroscience Institute, West Virginia University School of Medicine) M Milos R. Filipovic (Leibniz Institute for Analytical Sciences) B Benjamin C. Orsburn (Department of Physiology, Pharmacology, & Therapeutics, Johns Hopkins University School of Medicine) S Solomon H. Snyder (Department of Physiology, Pharmacology, & Therapeutics, Johns Hopkins University School of Medicine) A Andrew A. Pieper (Department of Psychiatry, Case Western Reserve University) B Bindu D. Paul (Department of Physiology, Pharmacology, & Therapeutics, Johns Hopkins University School of Medicine)

Abstract

Cystathionine γ-lyase (CSE), the enzyme responsible for neuronal cysteine and hydrogen sulfide production, is dysregulated in aging and neurodegenerative diseases including Alzheimer’s disease and Huntington’s disease, both marked by cognitive decline in addition to motor deficits. To determine whether CSE loss directly causes cognitive decline, we genetically ablated CSE in mice. This loss was sufficient to induce oxidative damage, compromise blood–brain barrier integrity, impair neurogenesis and neurotrophin signaling, and elicit cognitive deficits. Global proteomic analysis further revealed molecular alterations that contribute to impaired neurogenesis. Our findings establish CSE as an essential guardian of homeostatic brain health and identify it as a potential therapeutic target for neurodegenerative disorders.

Article Details

Volume / Issue Vol. 122, Issue 52
Published December 30, 2025
ISSN 0027-8424
Publisher National Academy of Sciences

Authors (17)

S

Suwarna Chakraborty

Department of Physiology, Pharmacology, & Therapeutics, Johns Hopkins University School of Medicine

S

Sunil Jamuna Tripathi

Department of Physiology, Pharmacology, & Therapeutics, Johns Hopkins University School of Medicine

E

Edwin Vázquez-Rosa

Department of Psychiatry, Case Western Reserve University

K

Kalyani Chaubey

Department of Psychiatry, Case Western Reserve University

H

Hisashi Fujioka

Cryo-Electron Microscopy Core Facility, Case Western Reserve University School of Medicine

E

Emiko Miller

Department of Psychiatry, Case Western Reserve University

R

Richa Tyagi

The Solomon H. Snyder Department of Neuroscience, Johns Hopkins University School of Medicine

T

Thibaut Vignane

Leibniz Institute for Analytical Sciences

S

Sudarshana M. Sharma

Department of Biochemistry and Molecular Biology and Hollings Cancer Center, Medical University of South Carolina

B

Bobby Thomas

Darby Children’s Research Institute, Medical University of South Carolina

Z

Zachary M. Weil

Department of Neuroscience, Rockefeller Neuroscience Institute, West Virginia University School of Medicine

R

Randy J. Nelson

Department of Neuroscience, Rockefeller Neuroscience Institute, West Virginia University School of Medicine

M

Milos R. Filipovic

Leibniz Institute for Analytical Sciences

B

Benjamin C. Orsburn

Department of Physiology, Pharmacology, & Therapeutics, Johns Hopkins University School of Medicine

S

Solomon H. Snyder

Department of Physiology, Pharmacology, & Therapeutics, Johns Hopkins University School of Medicine

A

Andrew A. Pieper

Department of Psychiatry, Case Western Reserve University

B

Bindu D. Paul

Department of Physiology, Pharmacology, & Therapeutics, Johns Hopkins University School of Medicine