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Proc Natl Acad Sci U S A ; 113(30): E4387-96, 2016 07 26.
Artigo em Inglês | MEDLINE | ID: mdl-27407143

RESUMO

The LIM-homeodomain transcription factors Lmx1a and Lmx1b play critical roles during the development of midbrain dopaminergic progenitors, but their functions in the adult brain remain poorly understood. We show here that sustained expression of Lmx1a and Lmx1b is required for the survival of adult midbrain dopaminergic neurons. Strikingly, inactivation of Lmx1a and Lmx1b recreates cellular features observed in Parkinson's disease. We found that Lmx1a/b control the expression of key genes involved in mitochondrial functions, and their ablation results in impaired respiratory chain activity, increased oxidative stress, and mitochondrial DNA damage. Lmx1a/b deficiency caused axonal pathology characterized by α-synuclein(+) inclusions, followed by a progressive loss of dopaminergic neurons. These results reveal the key role of these transcription factors beyond the early developmental stages and provide mechanistic links between mitochondrial dysfunctions, α-synuclein aggregation, and the survival of dopaminergic neurons.


Assuntos
Neurônios Dopaminérgicos/metabolismo , Proteínas com Homeodomínio LIM/genética , Mesencéfalo/metabolismo , Mitocôndrias/metabolismo , Fatores de Transcrição/genética , Animais , Sobrevivência Celular/genética , Dano ao DNA , Regulação da Expressão Gênica no Desenvolvimento , Células HEK293 , Humanos , Proteínas com Homeodomínio LIM/deficiência , Camundongos Endogâmicos C57BL , Camundongos Knockout , Camundongos Transgênicos , Mitocôndrias/genética , Estresse Oxidativo , Agregação Patológica de Proteínas , Fatores de Transcrição/deficiência , alfa-Sinucleína/metabolismo
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