Literature DB >> 9179155

Mitochondrial dysfunction with myoclonus epilepsy and ragged-red fibers point mutation in nerve, muscle, and adipose tissue of a patient with multiple symmetric lipomatosis.

M Naumann1, R Kiefer, K V Toyka, C Sommer, P Seibel, H Reichmann.   

Abstract

We report a 64-year-old man presenting with multiple symmetric lipomatosis (MSL) and mitochondrial encephalomyoneuropathy. The diagnosis of a mitochondrial cytopathy was based on the typical clinical symptoms and signs, including chronic progressive external ophthalmoplegia, hearing impairment, cerebellar ataxia, proximal myopathy, and polyneuropathy, and on molecular genetic and histological examinations. As a unique finding, the A-->G(8344) myoclonus epilepsy and ragged-red fibers point mutation was found in peripheral nerve, muscle, and adipose tissue. Muscle biopsy revealed multiple ragged-red fibers and other morphological signs of a mitochondrial myopathy. Sural nerve biopsy demonstrated a mixed axonal and demyelinating neuropathy with extensive loss of myelinated fibers and conspicuous onion bulb formations, as well as structural mitochondrial abnormalities on electron microscopy. These findings clearly demonstrate mitochondrial dysfunction in muscle, adipose tissue, and for the first time also in nervous tissue of an MSL patient, and strongly support the concept of mitochondrial cytopathy as one of the possible causes of multiple symmetric lipomatosis.

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Year:  1997        PMID: 9179155     DOI: 10.1002/(sici)1097-4598(199707)20:7<833::aid-mus7>3.0.co;2-8

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


  5 in total

1.  Mitochondrial DNA mutations in multiple symmetric lipomatosis.

Authors:  T Klopstock; M Naumann; P Seibel; B Shalke; K Reiners; H Reichmann
Journal:  Mol Cell Biochem       Date:  1997-09       Impact factor: 3.396

Review 2.  Mitochondrial biogenesis: a therapeutic target for neurodevelopmental disorders and neurodegenerative diseases.

Authors:  Martine Uittenbogaard; Anne Chiaramello
Journal:  Curr Pharm Des       Date:  2014       Impact factor: 3.116

3.  Mitochondrial Toxicity Associated with Nucleoside Reverse Transcriptase Inhibitor Therapy.

Authors:  Cecilia M. Shikuma; Bruce Shiramizu
Journal:  Curr Infect Dis Rep       Date:  2001-12       Impact factor: 3.725

4.  Mitochondrial DNA mutation load in a family with the m.8344A>G point mutation and lipomas: a case study.

Authors:  Tina Dysgaard Jeppesen; Noor Al-Hashimi; Morten Duno; Flemming Wibrand; Grete Andersen; John Vissing
Journal:  Clin Case Rep       Date:  2017-11-02

5.  Lipomatosis Incidence and Characteristics in an Italian Cohort of Mitochondrial Patients.

Authors:  Olimpia Musumeci; Emanuele Barca; Costanza Lamperti; Serenella Servidei; Giacomo Pietro Comi; Maurizio Moggio; Tiziana Mongini; Gabriele Siciliano; Massimiliano Filosto; Elena Pegoraro; Guido Primiano; Dario Ronchi; Liliana Vercelli; Daniele Orsucci; Luca Bello; Massimo Zeviani; Michelangelo Mancuso; Antonio Toscano
Journal:  Front Neurol       Date:  2019-02-27       Impact factor: 4.086

  5 in total

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