Literature DB >> 29350304

Defective mitochondrial ATPase due to rare mtDNA m.8969G>A mutation-causing lactic acidosis, intellectual disability, and poor growth.

Pirjo Isohanni1,2, Christopher J Carroll3, Christopher B Jackson3, Max Pohjanpelto3, Tuula Lönnqvist4, Anu Suomalainen3,5.   

Abstract

Mutations in mitochondrial ATP synthase 6 (MT-ATP6) are a frequent cause of NARP (neurogenic muscle weakness, ataxia, and retinitis pigmentosa) or Leigh syndromes, especially a point mutation at nucleotide position 8993. M.8969G>A is a rare MT-ATP6 mutation, previously reported only in three individuals, causing multisystem disorders with mitochondrial myopathy, lactic acidosis, and sideroblastic anemia or IgA nephropathy. We present two siblings with the m.8969G>A mutation and a novel, substantially milder phenotype with lactic acidosis, poor growth, and intellectual disability. Our findings expand the phenotypic spectrum and show that mtDNA mutations should be taken account also with milder, stable phenotypes.

Entities:  

Keywords:  Intellectual disability; Lactic acidosis; Mitochondrial DNA; Mitochondrial diseases

Mesh:

Substances:

Year:  2018        PMID: 29350304     DOI: 10.1007/s10048-018-0537-9

Source DB:  PubMed          Journal:  Neurogenetics        ISSN: 1364-6745            Impact factor:   2.660


  15 in total

1.  A new mitochondrial disease associated with mitochondrial DNA heteroplasmy.

Authors:  I J Holt; A E Harding; R K Petty; J A Morgan-Hughes
Journal:  Am J Hum Genet       Date:  1990-03       Impact factor: 11.025

2.  Adult-onset ataxia and polyneuropathy caused by mitochondrial 8993T-->C mutation.

Authors:  Maria T Rantamäki; Heidi K Soini; Saara M Finnilä; Kari Majamaa; Bjarne Udd
Journal:  Ann Neurol       Date:  2005-08       Impact factor: 10.422

3.  A second missense mutation in the mitochondrial ATPase 6 gene in Leigh's syndrome.

Authors:  D D de Vries; B G van Engelen; F J Gabreëls; W Ruitenbeek; B A van Oost
Journal:  Ann Neurol       Date:  1993-09       Impact factor: 10.422

4.  A novel mitochondrial ATPase 6 point mutation in familial bilateral striatal necrosis.

Authors:  D Thyagarajan; S Shanske; M Vazquez-Memije; D De Vivo; S DiMauro
Journal:  Ann Neurol       Date:  1995-09       Impact factor: 10.422

5.  Heteroplasmic mtDNA mutation (T----G) at 8993 can cause Leigh disease when the percentage of abnormal mtDNA is high.

Authors:  Y Tatuch; J Christodoulou; A Feigenbaum; J T Clarke; J Wherret; C Smith; N Rudd; R Petrova-Benedict; B H Robinson
Journal:  Am J Hum Genet       Date:  1992-04       Impact factor: 11.025

6.  Fatal neonatal lactic acidosis caused by a novel de novo mitochondrial G7453A tRNA-Serine ((UCN)) mutation.

Authors:  Alexandra Götz; Pirjo Isohanni; Brita Liljeström; Jaana Rummukainen; Kari Nikolajev; Eila Herrgård; Sanna Marjavaara; Anu Suomalainen
Journal:  Pediatr Res       Date:  2012-03-27       Impact factor: 3.756

7.  A novel mitochondrial ATP6 frameshift mutation causing isolated complex V deficiency, ataxia and encephalomyopathy.

Authors:  Christopher B Jackson; Dagmar Hahn; Barbara Schröter; Uwe Richter; Brendan J Battersby; Thomas Schmitt-Mechelke; Paula Marttinen; Jean-Marc Nuoffer; André Schaller
Journal:  Eur J Med Genet       Date:  2017-04-13       Impact factor: 2.708

8.  Mitochondrial myopathy, lactic acidosis, and sideroblastic anemia (MLASA) plus associated with a novel de novo mutation (m.8969G>A) in the mitochondrial encoded ATP6 gene.

Authors:  Lindsay C Burrage; Sha Tang; Jing Wang; Taraka R Donti; Magdalena Walkiewicz; J Michael Luchak; Li-Chieh Chen; Eric S Schmitt; Zhiyv Niu; Rodrigo Erana; Jill V Hunter; Brett H Graham; Lee-Jun Wong; Fernando Scaglia
Journal:  Mol Genet Metab       Date:  2014-06-30       Impact factor: 4.797

9.  SDHA mutation with dominant transmission results in complex II deficiency with ocular, cardiac, and neurologic involvement.

Authors:  Carolina Courage; Christopher B Jackson; Dagmar Hahn; Liliya Euro; Jean-Marc Nuoffer; Sabina Gallati; André Schaller
Journal:  Am J Med Genet A       Date:  2016-09-28       Impact factor: 2.802

10.  Identification of G8969>A in mitochondrial ATP6 gene that severely compromises ATP synthase function in a patient with IgA nephropathy.

Authors:  Shuzhen Wen; Katarzyna Niedzwiecka; Weiwei Zhao; Shutian Xu; Shaoshan Liang; Xiaodong Zhu; Honglang Xie; Déborah Tribouillard-Tanvier; Marie-France Giraud; Caihong Zeng; Alain Dautant; Róża Kucharczyk; Zhihong Liu; Jean-Paul di Rago; Huimei Chen
Journal:  Sci Rep       Date:  2016-11-04       Impact factor: 4.379

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  3 in total

1.  Recurrent heteroplasmy for the MT-ATP6 p.Ser148Asn (m.8969G>A) mutation in patients with syndromic congenital sideroblastic anemia of variable clinical severity.

Authors:  Simon Berhe; Matthew M Heeney; Dean R Campagna; John F Thompson; Eric J White; Tristen Ross; Roy W A Peake; Jeffery D Hanrahan; Vilmarie Rodriguez; Deborah L Renaud; Mrinal S Patnaik; Eugenia Chang; Sylvia S Bottomley; Mark D Fleming
Journal:  Haematologica       Date:  2018-07-13       Impact factor: 9.941

2.  Reply to 'Letter to Editor by Finsterer J and Zarrouk-Mahjoub S: Phenotypic manifestations of the m.8969G>A variant'.

Authors:  Pirjo Isohanni; Christopher J Carroll; Christopher B Jackson; Max Pohjanpelto; Tuula Lönnqvist; Anu Suomalainen
Journal:  Neurogenetics       Date:  2018-02-26       Impact factor: 2.660

3.  Phenotypic manifestations of the m.8969G>A variant.

Authors:  Josef Finsterer; Sinda Zarrouk-Mahjoub
Journal:  Neurogenetics       Date:  2018-02-26       Impact factor: 2.660

  3 in total

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