TY - JOUR IS - 50 PB - National Academy of Sciences Y1 - 2017/12/12/ A1 - Rott, R A1 - Szargel, R A1 - Shani, V A1 - Hamza, H A1 - Savyon, M A1 - Abd Elghani, F A1 - Bandopadhyay, R A1 - Engelender, S SP - 13176 UR - https://doi.org/10.1073/pnas.1704351114 KW - Parkinson?s disease KW - ?-synuclein KW - SUMOylatio KW - nubiquitination KW - aggregation TI - SUMOylation and ubiquitination reciprocally regulate ?-synuclein degradation and pathological aggregation N2 - ?-Synuclein accumulation is a pathological hallmark of Parkinson?s disease (PD). Ubiquitinated ?-synuclein is targeted to proteasomal or lysosomal degradation. Here, we identify SUMOylation as a major mechanism that counteracts ubiquitination by different E3 ubiquitin ligases and regulates ?-synuclein degradation. We report that PIAS2 promotes SUMOylation of ?-synuclein, leading to a decrease in ?-synuclein ubiquitination by SIAH and Nedd4 ubiquitin ligases, and causing its accumulation and aggregation into inclusions. This was associated with an increase in ?-synuclein release from the cells. A SUMO E1 inhibitor, ginkgolic acid, decreases ?-synuclein levels by relieving the inhibition exerted on ?-synuclein proteasomal degradation. ?-Synuclein disease mutants are more SUMOylated compared with the wild-type protein, and this is associated with increased aggregation and inclusion formation. We detected a marked increase in PIAS2 expression along with SUMOylated ?-synuclein in PD brains, providing a causal mechanism underlying the up-regulation of ?-synuclein SUMOylation in the disease. We also found a significant proportion of Lewy bodies in nigral neurons containing SUMO1 and PIAS2. Our observations suggest that SUMOylation of ?-synuclein by PIAS2 promotes ?-synuclein aggregation by two mutually reinforcing mechanisms. First, it has a direct proaggregatory effect on ?-synuclein. Second, SUMOylation facilitates ?-synuclein aggregation by blocking its ubiquitin-dependent degradation pathways and promoting its accumulation. Therefore, inhibitors of ?-synuclein SUMOylation provide a strategy to reduce ?-synuclein levels and possibly aggregation in PD. VL - 114 N1 - This version is the author accepted manuscript. For information on re-use, please refer to the publisher?s terms and conditions. SN - 0027-8424 ID - discovery10032511 AV - public JF - Proceedings of the National Academy of Sciences of the United States of America EP - 13181 ER -