Evidence map›Paper›PMID 26207760›Full record

Observational studyPloS one2015

Diagnostic Algorithm for Glycogenoses and Myoadenylate Deaminase Deficiency Based on Exercise Testing Parameters: A Prospective Study.

Fabrice Rannou, Arnaud Uguen, Virginie Scotet, Cédric Le Maréchal, Odile Rigal, Pascale Marcorelles, Eric Gobin, Jean-Luc Carré, Fabien Zagnoli, Marie-Agnès Giroux-Metges

Abstract readObservational Study
In one paragraph

Observational study in PloS one, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

2 citing papers in PubMed.

  1. Article
  2. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

10 authors.

Fabrice RannouPhysiology Department-EA 1274, CHRU Cavale Blanche, Brest, France.
Arnaud UguenPathology Department, CHRU Morvan, Brest, France.
Virginie ScotetInstitut National de la Santé et de la Recherche Médicale, UMR 1078, Brest, France.
Cédric Le MaréchalInstitut National de la Santé et de la Recherche Médicale, UMR 1078, Brest, France.
Odile RigalBiochemistry Department, Robert Debré Hospital-APHP, Paris, France.
Pascale MarcorellesPathology Department-EA 4685 LNB, CHRU Morvan, Brest, France.
Eric GobinPathology Department, CHRU Morvan, Brest, France.
Jean-Luc CarréBiochemistry Department, CHRU Cavale Blanche, Brest, France.
Fabien ZagnoliNeurology Department-EA 4685 LNB, Clermont-Tonnerre Armed Forces Hospital, Brest, France.
Marie-Agnès Giroux-MetgesPhysiology Department-EA 1274, CHRU Cavale Blanche, Brest, France.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

aimOur aim was to evaluate the accuracy of aerobic exercise testing to diagnose metabolic myopathies.

methodsFrom December 2008 to September 2012, all the consecutive patients that underwent both metabolic exercise testing and a muscle biopsy were prospectively enrolled. Subjects performed an incremental and maximal exercise testing on a cycle ergometer. Lactate, pyruvate, and ammonia concentrations were determined from venous blood samples drawn at rest, during exercise (50% predicted maximal power, peak exercise), and recovery (2, 5, 10, and 15 min). Biopsies from vastus lateralis or deltoid muscles were analysed using standard techniques (reference test). Myoadenylate deaminase (MAD) activity was determined using p-nitro blue tetrazolium staining in muscle cryostat sections. Glycogen storage was assessed using periodic acid-Schiff staining. The diagnostic accuracy of plasma metabolite levels to identify absent and decreased MAD activity was assessed using Receiver Operating Characteristic (ROC) curve analysis.

resultsThe study involved 51 patients. Omitting patients with glycogenoses (n = 3), MAD staining was absent in 5, decreased in 6, and normal in 37 subjects. Lactate/pyruvate at the 10th minute of recovery provided the greatest area under the ROC curves (AUC, 0.893 ± 0.067) to differentiate Abnormal from Normal MAD activity. The lactate/rest ratio at the 10th minute of recovery from exercise displayed the best AUC (1.0) for discriminating between Decreased and Absent MAD activities. The resulting decision tree achieved a diagnostic accuracy of 86.3%.

conclusionThe present algorithm provides a non-invasive test to accurately predict absent and decreased MAD activity, facilitating the selection of patients for muscle biopsy and target appropriate histochemical analysis.

Indexed as

AlgorithmsExercise TestAdolescentAdultAMP DeaminaseBiopsyExerciseFemaleGlycogen Storage DiseaseHumansMaleMiddle AgedMuscle, SkeletalPurine-Pyrimidine Metabolism, Inborn ErrorsYoung AdultAMP Deaminase

Identifiers

PMID26207760
PMCPMC4514803

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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.