Literature DB >> 28932990

Treatment Opportunities in Patients With Metabolic Myopathies.

Mette Cathrine Ørngreen1, John Vissing2.   

Abstract

Metabolic myopathies are disorders affecting utilization of carbohydrates or fat in the skeletal muscle. Adult patients with metabolic myopathies typically present with exercise-induced pain, contractures or stiffness, fatigue, and myoglobinuria. Symptoms are related to energy failure. Purpose of review In this review, the current treatment options, including exercise therapy, dietary treatment, pharmacological supplementation, gene transcription, and enzyme replacement therapy, are described. Recent findings Recognition of the metabolic block in the metabolic myopathies has started the development of new therapeutic options. Enzyme replacement therapy with rGAA has revolutionized treatment of early onset Pompe disease. Supplements of riboflavin, carnitine, and sucrose show promise in patients with respectively riboflavin-responsive multiple acyl-CoA dehydrogenase deficiency, primary carnitine deficiency, and McArdle disease. Treatment with citric acid cycle intermediates supply by triheptanoin seems promising in patients with glucogenoses, and studies are ongoing in patients with McArdle disease. Summary Treatment of metabolic myopathies primarily relies on avoiding precipitating factors and dietary supplements that bypass the metabolic block. Only a few of the used supplements are validated, and further studies are needed to define efficacious treatments. Further potential treatment targets are molecular therapies aimed at enzyme correction, such as chaperone therapy, gene therapy, gene expression therapy, and enzyme replacement therapies.

Entities:  

Keywords:  Diet therapy; Enzyme replacement therapy; Exercise therapy; Glycogenoses; Lipid metabolism disorders; Metabolic myopathies

Year:  2017        PMID: 28932990     DOI: 10.1007/s11940-017-0473-2

Source DB:  PubMed          Journal:  Curr Treat Options Neurol        ISSN: 1092-8480            Impact factor:   3.598


  84 in total

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2.  Long-term follow-up of bezafibrate treatment in patients with the myopathic form of carnitine palmitoyltransferase 2 deficiency.

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Journal:  N Engl J Med       Date:  2009-07-23       Impact factor: 91.245

4.  Effect of high-dose creatine therapy on symptoms of exercise intolerance in McArdle disease: double-blind, placebo-controlled crossover study.

Authors:  Matthias Vorgerd; Jochen Zange; Rudolf Kley; T Grehl; Anika Hüsing; Matthias Jäger; Klaus Müller; Rolf Schröder; Wilhelm Mortier; Klaus Fabian; Jean-Pierre Malin; Alwin Luttmann
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Journal:  Drugs       Date:  1987-06       Impact factor: 9.546

Review 6.  Pompe's disease.

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Journal:  Lancet       Date:  2008-10-11       Impact factor: 79.321

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Authors:  I Tein
Journal:  Semin Pediatr Neurol       Date:  1996-06       Impact factor: 1.636

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Journal:  Eur J Clin Pharmacol       Date:  1980-04       Impact factor: 2.953

10.  Safety and efficacy of exercise training in adults with Pompe disease: evalution of endurance, muscle strength and core stability before and after a 12 week training program.

Authors:  Linda E M van den Berg; Marein M Favejee; Stephan C A Wens; Michelle E Kruijshaar; Stephan F E Praet; Arnold J J Reuser; Johannes B J Bussmann; Pieter A van Doorn; Ans T van der Ploeg
Journal:  Orphanet J Rare Dis       Date:  2015-07-19       Impact factor: 4.123

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  6 in total

Review 1.  Myopathies Related to Glycogen Metabolism Disorders.

Authors:  Mark A Tarnopolsky
Journal:  Neurotherapeutics       Date:  2018-10       Impact factor: 7.620

2.  Medium branched chain fatty acids improve the profile of tricarboxylic acid cycle intermediates in mitochondrial fatty acid β-oxidation deficient cells: A comparative study.

Authors:  Anuradha Karunanidhi; Clinton Van't Land; Dhivyaa Rajasundaram; Mateus Grings; Jerry Vockley; Al-Walid Mohsen
Journal:  J Inherit Metab Dis       Date:  2022-02-02       Impact factor: 4.750

Review 3.  Pain Phenotypes in Rare Musculoskeletal and Neuromuscular Diseases.

Authors:  Anthony Tucker-Bartley; Jordan Lemme; Andrea Gomez-Morad; Nehal Shah; Miranda Veliu; Frank Birklein; Claudia Storz; Seward Rutkove; David Kronn; Alison M Boyce; Eduard Kraft; Jaymin Upadhyay
Journal:  Neurosci Biobehav Rev       Date:  2021-02-10       Impact factor: 9.052

Review 4.  Glutaric Acidemia, Pathogenesis and Nutritional Therapy.

Authors:  Qian Li; Chunlan Yang; Lijuan Feng; Yazi Zhao; Yong Su; Hong Liu; Hongkang Men; Yan Huang; Heinrich Körner; Xinming Wang
Journal:  Front Nutr       Date:  2021-12-15

5.  Exercise therapy in muscle diseases: open issues and future perspectives.

Authors:  Gabriele Siciliano; Erika Schirinzi; Costanza Simoncini; Giulia Ricci
Journal:  Acta Myol       Date:  2019-12-01

6.  Aerobic capacity and skeletal muscle characteristics in glycogen storage disease IIIa: an observational study.

Authors:  Philip J Hennis; Elaine Murphy; Rick I Meijer; Robin H Lachmann; Radha Ramachandran; Claire Bordoli; Gurinder Rayat; David J Tomlinson
Journal:  Orphanet J Rare Dis       Date:  2022-01-31       Impact factor: 4.123

  6 in total

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