Literature DB >> 28695670

Mitochondrial dysfunction and cerebral metabolic abnormalities in patients with mitochondrial encephalomyopathy subtypes: Evidence from proton MR spectroscopy and muscle biopsy.

Feng-Nan Niu1, Hai-Lan Meng2, Lei-Lei Chang2, Hong-Yan Wu1, Wei-Ping Li3, Ren-Yuan Liu2, Hui-Ting Wang3, Bing Zhang3, Yun Xu2.   

Abstract

AIMS: Accumulated evidence indicates that cerebral metabolic features, evaluated by proton magnetic resonance spectroscopy (1 H-MRS), are sensitive to early mitochondrion dysfunction associated with mitochondrial encephalomyopathy (ME). The metabolite ratios of lactate (lac)/Cr, N-acetyl aspartate (NAA)/creatine (Cr), total choline (tCho)/Cr, and myoinositol (mI)/Cr are measured in the infarct-like lesions by 1 H-MRS and may reveal metabolic changes associated with ME. However, the application of this molecular imaging technique in the investigation of the pathology of ME subtypes is unknown.
METHODS: In this study, cerebral metabolic features of pathologically diagnosed ME cases, that is, 19 mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS); nine chronic progressive external ophthalmoplegia (CPEO); and 23 healthy controls, were investigated using 1 H-MRS. Receiver operating characteristics (ROC) analysis was used to evaluate the diagnostic power of the cerebral metabolites. Histochemical evaluation was carried out on muscle tissues derived from biopsy to assess the abnormal mitochondrial proliferation. The association between cerebral metabolic and mitochondrial cytopathy was examined by correlation analysis.
RESULTS: Patients with MELAS or CPEO exhibited a significantly higher Lac/Cr ratio and a lower NAA/Cr ratio compared with controls. The ROC curve of Lac/Cr ratio indicated prominent discrimination between MELAS or CPEO and healthy control subjects, whereas the NAA/Cr ratio may present diagnostic power in the distinction of MELAS from CPEO. Lower NAA/Cr ratio was associated with higher Lac/Cr in MELAS, but not in CPEO. Furthermore, higher ragged-red fibers (RRFs) percentages were associated with elevated Lac/Cr and reduced NAA/Cr ratios, notably in MELAS. This association was not noted in the case of mI/Cr ratio.
CONCLUSIONS: Mitochondrial cytopathy (lactic acidosis and RRFs on muscle biopsy) was associated with neuronal viability but not glial proliferation, notably in MELAS. Mitochondrial neuronopathy and neuronal vulnerability are considered significant causes in the pathogenesis of MELAS, particularly with regard to stroke-like episodes.
© 2017 John Wiley & Sons Ltd.

Entities:  

Keywords:  zzm321990CPEOzzm321990; 1H-MRS; MELAS; cerebral metabolic abnormalities; mitochondrial cytopathy

Mesh:

Year:  2017        PMID: 28695670      PMCID: PMC6492760          DOI: 10.1111/cns.12714

Source DB:  PubMed          Journal:  CNS Neurosci Ther        ISSN: 1755-5930            Impact factor:   5.243


  29 in total

1.  Oxidative phosphorylation defect in the brains of carriers of the tRNAleu(UUR) A3243G mutation in a MELAS pedigree.

Authors:  F Dubeau; N De Stefano; B G Zifkin; D L Arnold; E A Shoubridge
Journal:  Ann Neurol       Date:  2000-02       Impact factor: 10.422

2.  Quantitative proton magnetic resonance spectroscopy of cerebral metabolic disturbances in patients with MELAS.

Authors:  E Wilichowski; P J Pouwels; J Frahm; F Hanefeld
Journal:  Neuropediatrics       Date:  1999-10       Impact factor: 1.947

3.  Magnetic resonance spectroscopy in patients with MELAS.

Authors:  Harald E Möller; Gerhard Kurlemann; Manfred Pützler; Dirk Wiedermann; Thorsten Hilbich; Barbara Fiedler
Journal:  J Neurol Sci       Date:  2004-12-15       Impact factor: 3.181

Review 4.  Adaptive responses of vertebrate neurons to anoxia--matching supply to demand.

Authors:  L T Buck; M E Pamenter
Journal:  Respir Physiol Neurobiol       Date:  2006-03-22       Impact factor: 1.931

Review 5.  Pathology of mitochondrial encephalomyopathies.

Authors:  Harvey B Sarnat; José Marín-García
Journal:  Can J Neurol Sci       Date:  2005-05       Impact factor: 2.104

6.  Evaluation of mitochondrial encephalomyopathy with lactic acidosis and stroke-like episodes with magnetic resonance imaging and proton magnetic resonance spectroscopy.

Authors:  Feng Feng; Hui You; Jing Gao; Xiao-Zhen Li; Chun-Ling Meng; Hong-Yi Sun; Zheng-Yu Jin; Yu-Pu Guo
Journal:  Chin Med Sci J       Date:  2006-12

7.  Mitochondrial disease criteria: diagnostic applications in children.

Authors:  E Morava; L van den Heuvel; F Hol; M C de Vries; M Hogeveen; R J Rodenburg; J A M Smeitink
Journal:  Neurology       Date:  2006-11-28       Impact factor: 9.910

8.  Serial brain imaging analysis of stroke-like episodes in MELAS.

Authors:  Hiromichi Ito; Kenji Mori; Masafumi Harada; Masako Minato; Etsuo Naito; Mayumi Takeuchi; Yasuhiro Kuroda; Shoji Kagami
Journal:  Brain Dev       Date:  2008-03-04       Impact factor: 1.961

9.  Cerebral lactic acidosis correlates with neurological impairment in MELAS.

Authors:  P Kaufmann; D C Shungu; M C Sano; S Jhung; K Engelstad; E Mitsis; X Mao; S Shanske; M Hirano; S DiMauro; D C De Vivo
Journal:  Neurology       Date:  2004-04-27       Impact factor: 9.910

10.  Proton MR spectroscopy of mitochondrial diseases: analysis of brain metabolic abnormalities and their possible diagnostic relevance.

Authors:  M Cristina Bianchi; Michela Tosetti; Roberta Battini; Maria L Manca; Michelangelo Mancuso; Giovanni Cioni; Raffaello Canapicchi; Gabriele Siciliano
Journal:  AJNR Am J Neuroradiol       Date:  2003 Nov-Dec       Impact factor: 3.825

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  2 in total

1.  Can MR spectroscopy and muscle biopsy findings be correlated with MELAS and CPEO?

Authors:  Josef Finsterer; Sinda Zarrouk-Mahjoub
Journal:  CNS Neurosci Ther       Date:  2017-08-29       Impact factor: 5.243

Review 2.  Molecular and neurological features of MELAS syndrome in paediatric patients: A case series and review of the literature.

Authors:  Lydia M Seed; Andrew Dean; Deepa Krishnakumar; Poe Phyu; Rita Horvath; Pooja Devi Harijan
Journal:  Mol Genet Genomic Med       Date:  2022-04-26       Impact factor: 2.473

  2 in total

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