| Literature DB >> 19945282 |
Sabrina Sacconi1, Eva Trevisson, Leonardo Salviati, Ségolène Aymé, Odile Rigal, Alberto Garcia Redondo, Michelangelo Mancuso, Gabriele Siciliano, Paola Tonin, Corrado Angelini, Karine Auré, Anne Lombès, Claude Desnuelle.
Abstract
Coenzyme Q(10) (CoQ(10)) deficiency has been associated with an increasing number of clinical phenotypes. Whereas primary CoQ(10) defects are related to mutations in ubiquinone biosynthetic genes, which are now being unraveled, and respond well to CoQ(10) supplementation, the etiologies, and clinical phenotypes related to secondary deficiencies are largely unknown. The purpose of this multicenter study was to evaluate the frequency of muscle CoQ(10) deficiency in a cohort of 76 patients presenting with clinically heterogeneous mitochondrial phenotypes which included myopathy among their clinical features. A reliable diagnostic tool based on HPLC quantification was employed to measure muscle CoQ(10) levels. A significant proportion of these patients (28 over 76) displayed CoQ(10) deficiency that was clearly secondary in nine patients, who harbored a pathogenic mutation of mitochondrial DNA. This study provides a rationale for future therapeutic trials on the effect of CoQ(10) supplementation in patients with mitochondrial diseases presenting with myopathy among clinical features.Entities:
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Year: 2009 PMID: 19945282 DOI: 10.1016/j.nmd.2009.10.014
Source DB: PubMed Journal: Neuromuscul Disord ISSN: 0960-8966 Impact factor: 4.296