Literature DB >> 8651651

Multiple mitochondrial DNA deletions in sporadic inclusion body myositis: a study of 56 patients.

F M Santorelli1, M Sciacco, K Tanji, S Shanske, T H Vu, V Golzi, R C Griggs, J R Mendell, A P Hays, T E Bertorini, A Pestronk, E Bonilla, S DiMauro.   

Abstract

Inclusion body myositis, a chronic inflammatory disorder, is the most common cause of myopathy in adults over the age of 50. Diagnosis is based on clinical features and distinctive morphological findings by both light and electron microscopy. The causes of inclusion body myositis are still unknown. Ultrastructural mitochondrial changes and ragged-red fibers are common in patients with sporadic inclusion body myositis, and multiple [correction of mutiple] mitochondrial DNA (mtDNA) deletions have been reported in 3 such patients, suggesting that mtDNA mutations may have a pathogenetic role. We studied 56 patients with sporadic inclusion body myositis, using a combination of clinical, morphological, biochemical, and molecular genetic analyses to determine the frequency and the distribution of mtDNA deletions. Using the polymerase chain reaction, we found multiple mtDNA deletions in 73% of patients, compared to 40% of normal age-matched control subjects and 47% of disease control subjects. The presence of deletions correlated with morphological evidence of ragged-red, cytochrome c oxidase-negative fibers, and with defects of complexes I and IV of the electron transport chain. Although aging may account for a proportion of mtDNA deletions in patients with sporadic inclusion body myositis and control subjects, mtDNA alterations may be accelerated in sporadic inclusion body myositis.

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Year:  1996        PMID: 8651651     DOI: 10.1002/ana.410390615

Source DB:  PubMed          Journal:  Ann Neurol        ISSN: 0364-5134            Impact factor:   10.422


  27 in total

1.  Muscle carnitine acetyltransferase and carnitine deficiency in a case of mitochondrial encephalomyopathy.

Authors:  B Melegh; L Seress; T Bedekovics; G Kispál; B Sümegi; K Trombitás; K Méhes
Journal:  J Inherit Metab Dis       Date:  1999-10       Impact factor: 4.982

Review 2.  Mitochondrial DNA analysis: polymorphisms and pathogenicity.

Authors:  P F Chinnery; N Howell; R M Andrews; D M Turnbull
Journal:  J Med Genet       Date:  1999-07       Impact factor: 6.318

3.  ANT1 is reduced in sporadic inclusion body myositis.

Authors:  E Barca; M Aguennouz; A Mazzeo; S Messina; A Toscano; G L Vita; S Portaro; D Parisi; C Rodolico
Journal:  Neurol Sci       Date:  2012-02-21       Impact factor: 3.307

4.  mtDNA single macrodeletions associated with myopathies: absence of haplogroup-related increased risk.

Authors:  A Goios; C Nogueira; C Pereira; L Vilarinho; A Amorim; L Pereira
Journal:  J Inherit Metab Dis       Date:  2005       Impact factor: 4.982

5.  Mitochondrial abnormalities and peripheral neuropathy in inflammatory myopathy, especially inclusion body myositis.

Authors:  J M Schröder; M Molnar
Journal:  Mol Cell Biochem       Date:  1997-09       Impact factor: 3.396

6.  Multiple mtDNA deletions: clinical and molecular correlations.

Authors:  F M Santorelli; G De Joanna; C Casali; A Tessa; G Siciliano; G A Amabile; F Pierelli; L Vilarinho; L Santoro
Journal:  J Inherit Metab Dis       Date:  2000-03       Impact factor: 4.982

7.  Follistatin Gene Therapy for Sporadic Inclusion Body Myositis Improves Functional Outcomes.

Authors:  Jerry R Mendell; Zarife Sahenk; Samiah Al-Zaidy; Louise R Rodino-Klapac; Linda P Lowes; Lindsay N Alfano; Katherine Berry; Natalie Miller; Mehmet Yalvac; Igor Dvorchik; Melissa Moore-Clingenpeel; Kevin M Flanigan; Kathleen Church; Kim Shontz; Choumpree Curry; Sarah Lewis; Markus McColly; Mark J Hogan; Brian K Kaspar
Journal:  Mol Ther       Date:  2017-03-06       Impact factor: 11.454

Review 8.  Polymyositis with mitochondrial pathology or atypical form of sporadic inclusion body myositis: case series and review of the literature.

Authors:  George K Papadimas; Charalampos Kokkinis; Sophia Xirou; Margarita Chrysanthou; Evangelia Kararizou; Constantinos Papadopoulos
Journal:  Rheumatol Int       Date:  2019-05-04       Impact factor: 2.631

9.  Detection and quantification of mitochondrial DNA deletions in individual cells by real-time PCR.

Authors:  Langping He; Patrick F Chinnery; Steve E Durham; Emma L Blakely; Theresa M Wardell; Gillian M Borthwick; Robert W Taylor; Douglass M Turnbull
Journal:  Nucleic Acids Res       Date:  2002-07-15       Impact factor: 16.971

10.  How citation distortions create unfounded authority: analysis of a citation network.

Authors:  Steven A Greenberg
Journal:  BMJ       Date:  2009-07-20
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