Age- and disease-dependent increase of the mitophagy marker phospho-ubiquitin in normal aging and Lewy body disease

Although exact causes of Parkinson disease (PD) remain enigmatic, mitochondrial dysfunction is increasingly appreciated as a key determinant of dopaminergic neuron susceptibility in both familial and sporadic PD. Two genes associated with recessive, early-onset PD encode the ubiquitin (Ub) kinase PI...

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Detalles Bibliográficos
Autores: Hou, Xu, Fiesel, Fabienne C., Truban, Dominika, Castanedes Casey, Monica, Lin, Wen-lang, Soto, Alexandra I., Tacik, Pawel, Rousseau, Linda G., Diehl, Nancy N., Heckman, Michael G., Lorenzo-Betancor, Oswaldo, Ferrer, Isidro (Ferrer Abizanda), Arbelo, José M., Steele, John C., Farrer, Matthew J., Cornejo-Olivas, Maria, Torres, Luis, Mata, Ignacio F., Graff-Radford, Neill R., Wszolek, Zbigniew K., Ross, Owen A., Murray, Melissa E., Dickson, Dennis W., Springer, Wolfdieter
Tipo de recurso: artículo
Estado:Versión aceptada para publicación
Fecha de publicación:2018
País:España
Institución:Varias* (Consorci de Biblioteques Universitáries de Catalunya, Centre de Serveis Científics i Acadèmics de Catalunya)
Repositorio:Recercat. Dipósit de la Recerca de Catalunya
OAI Identifier:oai:recercat.cat:2445/140074
Acceso en línea:https://hdl.handle.net/2445/140074
Access Level:acceso abierto
Palabra clave:Envelliment
Autofàgia
Mitocondris
Malaltia de Parkinson
Ubiqüitina
Aging
Autophagy
Mitochondria
Parkinson's disease
Ubiquitin
Descripción
Sumario:Although exact causes of Parkinson disease (PD) remain enigmatic, mitochondrial dysfunction is increasingly appreciated as a key determinant of dopaminergic neuron susceptibility in both familial and sporadic PD. Two genes associated with recessive, early-onset PD encode the ubiquitin (Ub) kinase PINK1 and the E3 Ub ligase PRKN/PARK2/Parkin, which together orchestrate a protective mitochondrial quality control (mitoQC) pathway. Upon stress, both enzymes cooperatively identify and decorate damaged mitochondria with phosphorylated poly-Ub (p-S65-Ub) chains. This specific label is subsequently recognized by autophagy receptors that further facilitate mitochondrial degradation in lysosomes (mitophagy). Here, we analyzed human post-mortem brain specimens and identified distinct pools of p-S65-Ub-positive structures that partially colocalized with markers of mitochondria, autophagy, lysosomes and/or granulovacuolar degeneration bodies. We further quantified levels and distribution of the 'mitophagy tag' in 2 large cohorts of brain samples from normal aging and Lewy body disease (LBD) cases using unbiased digital pathology. Somatic p-S65-Ub structures independently increased with age and disease in distinct brain regions and enhanced levels in LBD brain were age- and Braak tangle stage-dependent. Additionally, we observed significant correlations of p-S65-Ub with LBs and neurofibrillary tangle levels in disease. The degree of co-existing p-S65-Ub signals and pathological PD hallmarks increased in the pre-mature stage, but decreased in the late stage of LB or tangle aggregation. Altogether, our study provides further evidence for a potential pathogenic overlap among different forms of PD and suggests that p-S65-Ub can serve as a biomarker for mitochondrial damage in aging and disease.