Targeting MDM2 affects spastin protein levels and functions: implications for HSP treatment

Abstract Spastin is a microtubule (MT) severing enzyme that regulates several cell functions associated with MT dynamics. A reduction in spastin protein levels is responsible for approximately 40% of cases of Hereditary Spastic Paraplegia (HSP), a neurodegenerative disease. Currently, there is no cu...

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Main Authors: Francesca Sardina, Federica Polverino, Sonia Valentini, Claudia Carsetti, Elisabetta Falvo, Giada Tisci, Silvia Soddu, Fabiola Moretti, Alessandro Paiardini, Cinzia Rinaldo
Format: Article
Language:English
Published: Nature Publishing Group 2025-02-01
Series:Cell Death Discovery
Online Access:https://doi.org/10.1038/s41420-025-02333-y
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Summary:Abstract Spastin is a microtubule (MT) severing enzyme that regulates several cell functions associated with MT dynamics. A reduction in spastin protein levels is responsible for approximately 40% of cases of Hereditary Spastic Paraplegia (HSP), a neurodegenerative disease. Currently, there is no cure for HSP but strategies to induce a recovery of spastin levels are emerging as potential therapeutic approaches. Here, we show that MDM2 interacts with spastin MT-interacting and trafficking (MIT) domain. By biochemical and functional experiments, we demonstrate that MDM2 binds spastin and regulates its levels in a post-transcriptional manner independently of the E3 ubiquitin ligase activity. Of relevance, treatment of spastin-deficient cells with the MDM2 inhibitor Nutlin-3a can restore spastin levels and functions, such as cytokinetic abscission and sorting of transferrin receptor. These findings identify MDM2 as a novel interactor of spastin and a potential druggable regulator of its protein levels.
ISSN:2058-7716