Literature DB >> 33715266

Novel NDUFA12 variants are associated with isolated complex I defect and variable clinical manifestation.

Alessandra Torraco1, Alessia Nasca2, Daniela Verrigni1, Alessandra Pennisi3, Maha S Zaki4, Giorgia Olivieri5, Zahra Assouline3, Diego Martinelli5, Reza Maroofian6, Teresa Rizza1, Michela Di Nottia1, Federica Invernizzi2, Eleonora Lamantea2, Daniela Longo7, Henry Houlden6, Holger Prokisch8,9, Agnès Rötig3, Carlo Dionisi-Vici5, Enrico Bertini1, Daniele Ghezzi2,10, Rosalba Carrozzo1, Daria Diodato1.   

Abstract

Isolated biochemical deficiency of mitochondrial complex I is the most frequent signature among mitochondrial diseases and is associated with a wide variety of clinical symptoms. Leigh syndrome represents the most frequent neuroradiological finding in patients with complex I defect and more than 80 monogenic causes have been involved in the disease. In this report, we describe seven patients from four unrelated families harboring novel NDUFA12 variants, with six of them presenting with Leigh syndrome. Molecular genetic characterization was performed using next-generation sequencing combined with the Sanger method. Biochemical and protein studies were achieved by enzymatic activities, blue native gel electrophoresis, and western blot analysis. All patients displayed novel homozygous mutations in the NDUFA12 gene, leading to the virtual absence of the corresponding protein. Surprisingly, despite the fact that in none of the analyzed patients, NDUFA12 protein was detected, they present a different onset and clinical course of the disease. Our report expands the array of genetic alterations in NDUFA12 and underlines phenotype variability associated with NDUFA12 defect.
© 2021 Wiley Periodicals LLC.

Entities:  

Keywords:  Leigh syndrome; NADH ubiquinone oxidoreductase; NDUFA12; mitochondrial disease

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Year:  2021        PMID: 33715266     DOI: 10.1002/humu.24195

Source DB:  PubMed          Journal:  Hum Mutat        ISSN: 1059-7794            Impact factor:   4.878


  2 in total

1.  Biallelic Loss-of-Function NDUFA12 Variants Cause a Wide Phenotypic Spectrum from Leigh/Leigh-Like Syndrome to Isolated Optic Atrophy.

Authors:  Francesca Magrinelli; Elisa Cali; Vinícius Lopes Braga; Uluç Yis; Hoda Tomoum; Hanan Shamseldin; Julian Raiman; Christoph Kernstock; Flávio Moura Rezende Filho; Orlando Graziani Povoas Barsottini; Robert W Taylor; Elsebet Østergaard; Abdullah Tamim; Karin Schäferhoff; Juliana Maria Ferraz Sallum; Maha S Zaki; Fernando Kok; Kailash P Bhatia; Bernd Wissinger; Kate Sergeant; Tobias B Haack; Rita Horvath; Semra Hiz; Fowzan S Alkuraya; Henry Houlden; José Luiz Pedroso; Reza Maroofian
Journal:  Mov Disord Clin Pract       Date:  2022-01-03

Review 2.  Accessory Subunits of the Matrix Arm of Mitochondrial Complex I with a Focus on Subunit NDUFS4 and Its Role in Complex I Function and Assembly.

Authors:  Flora Kahlhöfer; Max Gansen; Volker Zickermann
Journal:  Life (Basel)       Date:  2021-05-19
  2 in total

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