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Epilepsy in MT-ATP6 - related mils/NARP: correlation of elettroclinical features with heteroplasmy.
Licchetta, Laura; Ferri, Lorenzo; La Morgia, Chiara; Zenesini, Corrado; Caporali, Leonardo; Lucia Valentino, Maria; Minardi, Raffaella; Fulitano, Daniela; Di Vito, Lidia; Mostacci, Barbara; Alvisi, Lara; Avoni, Patrizia; Liguori, Rocco; Tinuper, Paolo; Bisulli, Francesca; Carelli, Valerio.
Afiliación
  • Licchetta L; IRCCS Istituto delle Scienze Neurologiche di Bologna, Full Member of the ERN EpiCARE, Bologna, Italia.
  • Ferri L; Department of Biomedical and Neuromotor Sciences, University of Bologna, Bologna, Italia.
  • La Morgia C; Department of Biomedical and Neuromotor Sciences, University of Bologna, Bologna, Italia.
  • Zenesini C; IRCCS Istituto delle Scienze Neurologiche di Bologna, Full Member of the ERN EpiCARE, Bologna, Italia.
  • Caporali L; Department of Biomedical and Neuromotor Sciences, University of Bologna, Bologna, Italia.
  • Lucia Valentino M; IRCCS Istituto delle Scienze Neurologiche di Bologna, Full Member of the ERN EpiCARE, Bologna, Italia.
  • Minardi R; IRCCS Istituto delle Scienze Neurologiche di Bologna, Full Member of the ERN EpiCARE, Bologna, Italia.
  • Fulitano D; IRCCS Istituto delle Scienze Neurologiche di Bologna, Full Member of the ERN EpiCARE, Bologna, Italia.
  • Di Vito L; Department of Biomedical and Neuromotor Sciences, University of Bologna, Bologna, Italia.
  • Mostacci B; IRCCS Istituto delle Scienze Neurologiche di Bologna, Full Member of the ERN EpiCARE, Bologna, Italia.
  • Alvisi L; Neurology Unit, Rovigo, Italy.
  • Avoni P; IRCCS Istituto delle Scienze Neurologiche di Bologna, Full Member of the ERN EpiCARE, Bologna, Italia.
  • Liguori R; IRCCS Istituto delle Scienze Neurologiche di Bologna, Full Member of the ERN EpiCARE, Bologna, Italia.
  • Tinuper P; IRCCS Istituto delle Scienze Neurologiche di Bologna, Full Member of the ERN EpiCARE, Bologna, Italia.
  • Bisulli F; Department of Biomedical and Neuromotor Sciences, University of Bologna, Bologna, Italia.
  • Carelli V; IRCCS Istituto delle Scienze Neurologiche di Bologna, Full Member of the ERN EpiCARE, Bologna, Italia.
Ann Clin Transl Neurol ; 8(3): 704-710, 2021 03.
Article en En | MEDLINE | ID: mdl-33476484
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.
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Texto completo: 1 Colección: 01-internacional Banco de datos: MEDLINE Asunto principal: Enfermedad de Leigh / Retinitis Pigmentosa / Miopatías Mitocondriales / ATPasas de Translocación de Protón Mitocondriales / Ondas Encefálicas / Heteroplasmia Límite: Adult / Child, preschool / Female / Humans / Male / Middle aged Idioma: En Revista: Ann Clin Transl Neurol Año: 2021 Tipo del documento: Article País de afiliación: Italia

Texto completo: 1 Colección: 01-internacional Banco de datos: MEDLINE Asunto principal: Enfermedad de Leigh / Retinitis Pigmentosa / Miopatías Mitocondriales / ATPasas de Translocación de Protón Mitocondriales / Ondas Encefálicas / Heteroplasmia Límite: Adult / Child, preschool / Female / Humans / Male / Middle aged Idioma: En Revista: Ann Clin Transl Neurol Año: 2021 Tipo del documento: Article País de afiliación: Italia