| Literature DB >> 33476484 |
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.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