Literature DB >> 28981487

Oxygen consumption in platelets as an adjunct diagnostic method for pediatric mitochondrial disease.

Emil Westerlund1, Sigurður E Marelsson1, Johannes K Ehinger1, Fredrik Sjövall1, Saori Morota1, Eleonor Åsander Frostner1, Anders Oldfors2, Niklas Darin3, Johan Lundgren4, Magnus J Hansson1, Vineta Fellman4, Eskil Elmér1.   

Abstract

BackgroundDiagnosing mitochondrial disease (MD) is a challenge. In addition to genetic analyses, clinical practice is to perform invasive procedures such as muscle biopsy for biochemical and histochemical analyses. Blood cell respirometry is rapid and noninvasive. Our aim was to explore its possible role in diagnosing MD.MethodsBlood samples were collected from 113 pediatric patients, for whom MD was a differential diagnosis. A respiratory analysis model based on ratios (independent of mitochondrial specific content) was derived from a group of healthy controls and tested on the patients. The diagnostic accuracy of platelet respirometry was evaluated against routine diagnostic investigation.ResultsMD prevalence in the cohort was 16%. A ratio based on the respiratory response to adenosine diphosphate in the presence of complex I substrates had 96% specificity for disease and a positive likelihood ratio of 5.3. None of the individual ratios had sensitivity above 50%, but a combined model had 72% sensitivity.ConclusionNormal findings of platelet respirometry are not able to rule out MD, but pathological results make the diagnosis more likely and could strengthen the clinical decision to perform further invasive analyses. Our results encourage further study into the role of blood respirometry as an adjunct diagnostic tool for MD.

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Year:  2017        PMID: 28981487     DOI: 10.1038/pr.2017.250

Source DB:  PubMed          Journal:  Pediatr Res        ISSN: 0031-3998            Impact factor:   3.756


  22 in total

1.  Simplifying likelihood ratios.

Authors:  Steven McGee
Journal:  J Gen Intern Med       Date:  2002-08       Impact factor: 5.128

2.  Mitochondrial respiration in human viable platelets--methodology and influence of gender, age and storage.

Authors:  Fredrik Sjövall; Johannes K H Ehinger; Sigurður E Marelsson; Saori Morota; Eleonor Asander Frostner; Hiroyuki Uchino; Johan Lundgren; Einar Arnbjörnsson; Magnus J Hansson; Vineta Fellman; Eskil Elmér
Journal:  Mitochondrion       Date:  2012-11-16       Impact factor: 4.160

3.  Mitochondrial dysfunction in blood cells from amyotrophic lateral sclerosis patients.

Authors:  Johannes K Ehinger; Saori Morota; Magnus J Hansson; Gesine Paul; Eskil Elmér
Journal:  J Neurol       Date:  2015-04-18       Impact factor: 4.849

4.  Mitochondrial disorders: a proposal for consensus diagnostic criteria in infants and children.

Authors:  Nicole I Wolf; Jan A M Smeitink
Journal:  Neurology       Date:  2002-11-12       Impact factor: 9.910

5.  Diagnostic criteria for respiratory chain disorders in adults and children.

Authors:  F P Bernier; A Boneh; X Dennett; C W Chow; M A Cleary; D R Thorburn
Journal:  Neurology       Date:  2002-11-12       Impact factor: 9.910

6.  Oxygen consumption measurement in lymphocytes for the diagnosis of pediatric patients with oxidative phosphorylation diseases.

Authors:  R Artuch; C Colomé; A Playán; M J Alcaine; P Briones; J Montoya; M A Vilaseca; M Pineda
Journal:  Clin Biochem       Date:  2000-08       Impact factor: 3.281

Review 7.  Recognition, investigation and management of mitochondrial disease.

Authors:  James E Davison; Shamima Rahman
Journal:  Arch Dis Child       Date:  2017-06-24       Impact factor: 3.791

8.  Mitochondria, cognitive impairment, and Alzheimer's disease.

Authors:  M Mancuso; V Calsolaro; D Orsucci; C Carlesi; A Choub; S Piazza; G Siciliano
Journal:  Int J Alzheimers Dis       Date:  2009-07-06

Review 9.  The in-depth evaluation of suspected mitochondrial disease.

Authors:  Richard H Haas; Sumit Parikh; Marni J Falk; Russell P Saneto; Nicole I Wolf; Niklas Darin; Lee-Jun Wong; Bruce H Cohen; Robert K Naviaux
Journal:  Mol Genet Metab       Date:  2008-02-01       Impact factor: 4.797

10.  Noninvasive diagnostics of mitochondrial disorders in isolated lymphocytes with high resolution respirometry.

Authors:  Petr Pecina; Hana Houšťková; Tomáš Mráček; Alena Pecinová; Hana Nůsková; Markéta Tesařová; Hana Hansíková; Jan Janota; Jiří Zeman; Josef Houštěk
Journal:  BBA Clin       Date:  2014-10-01
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