| Literature DB >> 33385519 |
Manuella Maria Silva Santos1, Damián Gatica2, Jaqueline de Azêvedo Silva3, Sergio Crovella3, Daniel J Klionsky2, Marcos Antonio De Morais4.
Abstract
Mevalonate kinase deficiency (MKD) is an autosomal recessive disorder in humans that causes systemic autoinflammatory problems to children. Previously, we used a yeast model to show that MKD results in mitochondrial malfunctioning that may finally induce mitophagy. Here, we proved that MKD indeed induced general autophagy as well as mitophagy in yeast, but these mechanisms did not go to completion. Therefore, the limitation of mevalonate kinase activity produces dysfunctional mitochondria that might not be recycled, causing metabolic dysfunctions in the cells. Understanding this mechanism may provide a piece in solving the nonspecific autoinflammatory response puzzle observed in MKD patients.Entities:
Keywords: Autoinflammatory disorders; Autophagy; Erg12; Mevalonate kinase deficiency; Mitochondrial disorder; Mitophagy
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Year: 2020 PMID: 33385519 PMCID: PMC8011287 DOI: 10.1016/j.bbadis.2020.166053
Source DB: PubMed Journal: Biochim Biophys Acta Mol Basis Dis ISSN: 0925-4439 Impact factor: 5.187