Literature DB >> 9447609

On a dominantly inherited myopathy with tubular aggregates.

J J Martin1, C Ceuterick, G Van Goethem.   

Abstract

A 19-year-old patient presented with exercise-related myalgia, fatigue and elevated creatine kinase levels. Histology of a muscle biopsy was characterized by the presence of very large amounts of tubular aggregates. Both his father and paternal grandfather had elevated creatine kinase and large amounts of tubular aggregates in their muscle biopsies. The aggregates consisted of closely packed vesicles and tubules filled with electron-dense material or with one to several smaller tubules. Disorders with tubular aggregates in the muscle fibres such as hyperornithinaemia with gyrate atrophy of the retina, hypokalaemic periodic paralysis, hyperkalaemic periodic paralysis, myotonia congenita, alcoholism, osteomalacic myopathy etc. have been excluded. Tubular aggregates can be found in muscle disorders characterized by exercise-induced cramps, pain and stiffness. They also represent the predominant histological feature of some familial myopathies due to a yet unidentified genetic defect. In our family, there was male-to-male transmission, confirming dominant inheritance.

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Year:  1997        PMID: 9447609     DOI: 10.1016/s0960-8966(97)00119-3

Source DB:  PubMed          Journal:  Neuromuscul Disord        ISSN: 0960-8966            Impact factor:   4.296


  5 in total

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2.  Caveolin-1(-/-)- and caveolin-2(-/-)-deficient mice both display numerous skeletal muscle abnormalities, with tubular aggregate formation.

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4.  Molecular Characterization of Skeletal Muscle Dysfunction in Sigma 1 Receptor (Sigmar1) Knockout Mice.

Authors:  Richa Aishwarya; Chowdhury S Abdullah; Naznin S Remex; Shafiul Alam; Mahboob Morshed; Sadia Nitu; Brandon Hartman; Judy King; Mohammad Alfrad Nobel Bhuiyan; A Wayne Orr; Christopher G Kevil; Md Shenuarin Bhuiyan
Journal:  Am J Pathol       Date:  2021-10-25       Impact factor: 4.307

5.  Muscle MR imaging in tubular aggregate myopathy.

Authors:  Valeria Beltrame; Paolo Ortolan; Alessandro Coran; Riccardo Zanato; Matteo Gazzola; Annachiara Frigo; Luca Bello; Elena Pegoraro; Roberto Stramare
Journal:  PLoS One       Date:  2014-04-10       Impact factor: 3.240

  5 in total

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