Literature DB >> 33476484

Epilepsy in MT-ATP6 - related mils/NARP: correlation of elettroclinical features with heteroplasmy.

Laura Licchetta1,2, Lorenzo Ferri2, Chiara La Morgia1,2, Corrado Zenesini1, Leonardo Caporali1, Maria Lucia Valentino1,2, Raffaella Minardi1, Daniela Fulitano3, Lidia Di Vito1, Barbara Mostacci1, Lara Alvisi1,2, Patrizia Avoni1,2, Rocco Liguori1,2, Paolo Tinuper1,2, Francesca Bisulli1,2, Valerio Carelli1,2.   

Abstract

The study aims to characterize the epilepsy phenotype of maternally inherited Leigh's syndrome (MILS) and neuropathy, ataxia, retinitis pigmentosa (NARP) due to mutations in the mitochondrial ATP6 gene and to correlate electroclinical features with mutant heteroplasmy load (HL). We investigated 17 individuals with different phenotype, from asymptomatic carriers to MILS: 11 carried the m.8993T> G mutation, 5 the m.8993T> C and one the novel, de novo m.8858G> A mutation. Seizures occurred in 37.5% of patients, EEG abnormalities in 73%. We ranked clinical and EEG abnormalities severity and performed quantitative EEG to estimate Abnormality Ratio (AR) and Spectral Relative Power (SRP). Spearman's rho and Kruskal-Wallis test were used for correlation with heteroplasmy load (HL). HL correlated with disease severity (Rho = 0.63, P = 0.012) and was significantly higher in patients with seizures or EEG abnormalities (P = 0.014). HL correlated with EEG severity score only for the m.8993T> G (Rho = 0.73, P = 0.040), showing a trend toward a positive correlation with AR and delta SPR, irrespective of the mutation.
© 2021 The Authors. Annals of Clinical and Translational Neurology published by Wiley Periodicals LLC on behalf of American Neurological Association.

Entities:  

Keywords:  MT-ATP6 MT-ATP6; epilepsy; maternally inherited Leigh's syndrome (MILS); neuropathy, ataxia, retinitis pigmentosa (NARP); progressive myoclonic epilepsy (PME)

Year:  2021        PMID: 33476484     DOI: 10.1002/acn3.51259

Source DB:  PubMed          Journal:  Ann Clin Transl Neurol        ISSN: 2328-9503            Impact factor:   4.511


  3 in total

1.  Intractable Epilepsy in Maternally Inherited Leigh Syndrome (MILS) Due to the Sporadic Variant m.8993T>G in MT-ATP6: A Case Report.

Authors:  Josef Finsterer
Journal:  Cureus       Date:  2022-02-28

2.  Milder Phenotype of Homoplasmic Versus Heteroplasmic m.8344A>G Variant in the Same Family: A Case Report.

Authors:  Josef Finsterer; Sounira Mehri
Journal:  Cureus       Date:  2022-08-28

3.  Familial mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episode syndrome: Three case reports.

Authors:  Xiao Yang; Le-Jun Fu
Journal:  World J Clin Cases       Date:  2022-09-26       Impact factor: 1.534

  3 in total

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