Literature DB >> 2976895

Metabolic myopathy in canine muscle-type phosphofructokinase deficiency.

U Giger1, Z Argov, M Schnall, W J Bank, B Chance.   

Abstract

In vivo 31phosphorus nuclear magnetic resonance spectroscopy (P-NMR) of the anterior tibialis muscle was used to investigate the metabolic myopathy of inherited muscle-type phosphofructokinase (PFK) deficiency in four (homozygous) dogs who had mild exercise intolerance, rare muscle cramps, increased serum creatine kinase activity, but no myoglobinuria. During isometric muscle work induced by indirect electrical stimulation, and subsequent recovery, changes in the ratio of phosphocreatine (PCr) and inorganic phosphates (Pi) were comparable in muscle of PFK-deficient and normal dogs and indicated a large capacity for arobic oxidative phosphorylation in canine muscle. The progressive accumulation of sugar phosphates (PME) during graded exercise clearly demonstrated the glycolytic block in PFK-deficient dogs. During a muscle contracture, induced by acute muscle stimulation, PFK-deficient muscle became completely depleted of PCr and ATP, accumulated large amounts of PME, and recovered very slowly. We conclude that PFK-deficient dogs have a metabolic myopathy that demonstrated some but not all the features recognized in the human disorder.

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Year:  1988        PMID: 2976895     DOI: 10.1002/mus.880111210

Source DB:  PubMed          Journal:  Muscle Nerve        ISSN: 0148-639X            Impact factor:   3.217


  5 in total

1.  A complex rearrangement in GBE1 causes both perinatal hypoglycemic collapse and late-juvenile-onset neuromuscular degeneration in glycogen storage disease type IV of Norwegian forest cats.

Authors:  John C Fyfe; Rebeccah L Kurzhals; Michelle G Hawkins; Ping Wang; Naoya Yuhki; Urs Giger; Thomas J Van Winkle; Mark E Haskins; Donald F Patterson; Paula S Henthorn
Journal:  Mol Genet Metab       Date:  2007-01-25       Impact factor: 4.797

2.  Hereditary phosphofructokinase deficiency in wachtelhunds.

Authors:  Anna Hillström; Harold Tvedten; André Rowe; Urs Giger
Journal:  J Am Anim Hosp Assoc       Date:  2011-02-10       Impact factor: 1.023

Review 3.  Preclinical Development of New Therapy for Glycogen Storage Diseases.

Authors:  Baodong Sun; Elizabeth D Brooks; Dwight D Koeberl
Journal:  Curr Gene Ther       Date:  2015       Impact factor: 4.391

Review 4.  Large animal models and new therapies for glycogen storage disease.

Authors:  Elizabeth D Brooks; Dwight D Koeberl
Journal:  J Inherit Metab Dis       Date:  2014-09-16       Impact factor: 4.982

5.  EHBP1L1 Frameshift Deletion in English Springer Spaniel Dogs with Dyserythropoietic Anemia and Myopathy Syndrome (DAMS) or Neonatal Losses.

Authors:  Sarah Østergård Jensen; Matthias Christen; Veronica Rondahl; Christopher T Holland; Vidhya Jagannathan; Tosso Leeb; Urs Giger
Journal:  Genes (Basel)       Date:  2022-08-26       Impact factor: 4.141

  5 in total

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