Gene delivery of SUMO1-derived peptide rescues neuronal degeneration and motor deficits in a mouse model of Parkinson’s disease.

Publication date: Jul 02, 2025

Developing α-synuclein aggregation inhibitors is challenging because its aggregation process involves several microscopic steps and heterogeneous intermediates. Previously, we identified a SUMO1-derived peptide, SUMO1(15-55), that exhibits tight binding to monomeric α-synuclein via SUMO-SUMO-interacting motif (SIM) interactions, and effectively blocks the initiation of aggregation and formation of toxic aggregates in vitro. In cellular and Drosophila models, SUMO1(15-55) was efficacious in protecting neuronal cells from α-synuclein-induced neurotoxicity and neuronal degeneration. Given the demonstrated ability of SUMO1(15-55) to sequester α-synuclein monomers thereby blocking oligomer formation, we sought to evaluate whether it could be equally effective against the aggregation-prone familial mutant α-synuclein-A53T. Herein, we show that SUMO1(15-55) selectively binds to monomeric α-synuclein-A53T, inhibits primary nucleation, and prevents the formation of structured protofibrils in vitro, thereby protecting neuronal cells from protofibril-induced cell death. We further demonstrate that larval feeding of a designed His-SUMO1(15-55) that exhibits enhanced sub-stoichiometric suppression of α-synuclein-A53T aggregation in vitro can ameliorate Parkinson’s disease (PD)-related symptoms in α-synuclein-A53T transgenic Drosophila models, while its rAAV-mediated gene delivery can relieve the PD-related histological and behavioral deficiencies in an rAAV-α-synuclein-A53T mouse PD model. Our findings suggest that gene delivery of His-SUMO1(15-55) may serve as a clinical therapy for a spectrum of α-synuclein-aggregation associated synucleinopathies.

Concepts Keywords
Drosophila alpha-Synuclein
Feeding alpha-Synuclein
Parkinson Animals
Sumo115 Disease Models, Animal
Toxic gene therapy
Gene Transfer Techniques
Genetic Therapy
Humans
Mice
mouse behavioral tests
Nerve Degeneration
Neurons
Parkinson Disease
Peptides
Peptides
recombinant adeno-associated virus
small ubiquitin-like modifier
SUMO-1 Protein
SUMO-1 Protein
α-synuclein-Α53Τ aggregation

Semantics

Type Source Name
disease MESH Parkinson’s disease
disease MESH synucleinopathies
disease MESH Disease Models Animal
disease MESH Nerve Degeneration
pathway KEGG Parkinson disease

Original Article

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