Chronically low NMNAT2 expression causes sub-lethal SARM1 activation and altered response to nicotinamide riboside in axons
Christina Antoniou, Andrea LoretoiD, Jonathan GilleyiD, Elisa Merlini, Giuseppe OrsomandoiD, Michael P. ColemaniD
Abstract
Nicotinamide mononucleotide adenylyltransferase 2 (NMNAT2) is an endogenous axon survival factor that maintains axon health by blocking activation of the downstream pro-degenerative protein SARM1 (sterile alpha and TIR motif containing protein 1). While complete absence of NMNAT2 in mice results in extensive axon truncation and perinatal lethality, the removal of SARM1 completely rescues these phenotypes. Reduced levels of NMNAT2 can be compatible with life, however they compromise axon development and survival. Mice born expressing sub-heterozygous levels of NMNAT2 remain overtly normal into old age but develop axonal defects in vivo and in vitro as well as behavioural phenotypes. Therefore, it is important to examine the effects of constitutively low NMNAT2 expression on SARM1 activation and disease susceptibility. Here we demonstrate that chronically low NMNAT2 levels reduce prenatal viability in mice in a SARM1-dependent manner and lead to sub-lethal SARM1 activation in morphologically intact axons of superior cervical ganglion (SCG) primary cultures. This is characterised by a depletion in NAD(P) and compromised neurite outgrowth. We also show that chronically low NMNAT2 expression reverses the NAD-enhancing effect of nicotinamide riboside (NR) in axons in a SARM1-dependent manner. These data indicate that low NMNAT2 levels can trigger sub-lethal SARM1 activation which is detectable at the molecular level and could predispose to human axonal disorders.
02 /Specification & Classification
Click any tag to browse matching papers- Citations
- 8
- FWCI
- 1.40
- Field percentile
- top 19%
- Organism
- Approach
Reproducibility ledger
Nobody has reported trying to reproduce this paper yet. Did you replicate an experiment or run the code? Your report saves the next lab a week.
Sign in to file a reproducibility report.
Related papers
- Macrophage depletion blocks congenital SARM1-dependent neuropathy
Caitlin B. Dingwall, Amy M. Strickland, … Jeffrey Milbrandt·Journal of Clinical InvestigationPeer-reviewed·Oct 26, 2022
- Nicotinic acid mononucleotide is an allosteric SARM1 inhibitor promoting axonal protection
Yo Sasaki, Jian Zhu, … Jeffrey Milbrandt·Experimental NeurologyPeer-reviewed·Aug 14, 2021
- NAD homeostasis maintained by NMNAT2 supports vesicular glycolysis and fuels fast axonal transport in distal axons of cortical glutamatergic neurons in mice
Sen Yang, Zhen‐Xian Niou, … Hui‐Chen Lu·bioRxiv·Feb 6, 2022
- Severe Biallelic Loss-of-function Mutations in Nicotinamide Mononucleotide Adenylyltransferase 2 (NMNAT2) in Two Fetuses with Fetal Akinesia Deformation Sequence
Marshall Lukacs, Jonathan Gilley, … Rolf W. Stottmann·Experimental NeurologyPeer-reviewed·May 25, 2019
- The NAD+ precursor NMN activates dSarm to trigger axon degeneration in Drosophila
Arnau Llobet Rosell, Maria Paglione, … Lukas Jakob Neukomm·eLifePeer-reviewed·Dec 7, 2022
- Suppressing phagocyte activation by overexpressing the phosphatidylserine lipase ABHD12 preserves sarmopathic nerves
Caitlin B. Dingwall, Yo Sasaki, … Jeffrey Milbrandt·iSciencePeer-reviewed·May 9, 2025