Mitochondrial disorders of the OXPHOS system

E Fernandez‐Vizarra, M Zeviani - FEBS letters, 2021 - Wiley Online Library
Mitochondrial disorders are among the most frequent inborn errors of metabolism, their
primary cause being the dysfunction of the oxidative phosphorylation system (OXPHOS) …

Spinocerebellar ataxia: an update

R Sullivan, WY Yau, E O'Connor, H Houlden - Journal of neurology, 2019 - Springer
Spinocerebellar ataxia (SCA) is a heterogeneous group of neurodegenerative ataxic
disorders with autosomal dominant inheritance. We aim to provide an update on the recent …

[HTML][HTML] Hereditary ataxias: overview

S Jayadev, TD Bird - Genetics in Medicine, 2013 - Elsevier
The hereditary ataxias are a highly heterogeneous group of disorders phenotypically
characterized by gait ataxia, incoordination of eye movements, speech, and hand …

ATP synthase diseases of mitochondrial genetic origin

A Dautant, T Meier, A Hahn… - Frontiers in …, 2018 - frontiersin.org
Devastating human neuromuscular disorders have been associated to defects in the ATP
synthase. This enzyme is found in the inner mitochondrial membrane and catalyzes the last …

Human diseases associated with defects in assembly of OXPHOS complexes

D Ghezzi, M Zeviani - Essays in biochemistry, 2018 - portlandpress.com
The structural biogenesis and functional proficiency of the multiheteromeric complexes
forming the mitochondrial oxidative phosphorylation system (OXPHOS) require the …

MT‐ATP6 mitochondrial disease variants: Phenotypic and biochemical features analysis in 218 published cases and cohort of 14 new cases

RD Ganetzky, C Stendel, EM McCormick… - Human …, 2019 - Wiley Online Library
Mitochondrial complex V (CV) generates cellular energy as adenosine triphosphate (ATP).
Mitochondrial disease caused by the m. 8993T> G pathogenic variant in the CV subunit …

Mitochondrial protein dysfunction in pathogenesis of neurological diseases

L Wang, Z Yang, X He, S Pu, C Yang, Q Wu… - Frontiers in Molecular …, 2022 - frontiersin.org
Mitochondria are essential organelles for neuronal function and cell survival. Besides the
well-known bioenergetics, additional mitochondrial roles in calcium signaling, lipid …

[HTML][HTML] Understanding structure, function, and mutations in the mitochondrial ATP synthase

T Xu, V Pagadala, DM Mueller - Microbial cell, 2015 - ncbi.nlm.nih.gov
The mitochondrial ATP synthase is a multimeric enzyme complex with an overall molecular
weight of about 600,000 Da. The ATP synthase is a molecular motor composed of two …

Spinocerebellar ataxias (SCAs) caused by common mutations

U Müller - neurogenetics, 2021 - Springer
The term SCA refers to a phenotypically and genetically heterogeneous group of autosomal
dominant spinocerebellar ataxias. Phenotypically they present as gait ataxia frequently in …

A novel mutation m.8561C>G in MT-ATP6/8 causing a mitochondrial syndrome with ataxia, peripheral neuropathy, diabetes mellitus, and hypergonadotropic …

L Kytövuori, J Lipponen, H Rusanen, T Komulainen… - Journal of …, 2016 - Springer
Defects in the respiratory chain or mitochondrial ATP synthase (complex V) result in
mitochondrial dysfunction that is an important cause of inherited neurological disease. Two …