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Movement disorders : official journal of the Movement Disorder Society · 2014

Myoclonus in mitochondrial disorders.

СтатьяПоддержка исследования: вне правительства СШАНазвание и аннотация: Перевод пока не подготовлен
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Mancuso Michelangelo, Orsucci Daniele, Angelini Corrado, Bertini Enrico, Catteruccia Michela, Pegoraro Elena, Carelli Valerio, Valentino Maria L, Comi Giacomo P, Minetti Carlo, Bruno Claudio, Moggio Maurizio, Ienco Elena Caldarazzo, Mongini Tiziana, Vercelli Liliana, Primiano Guido, Servidei Serenella, Tonin Paola, Scarpelli Mauro, Toscano Antonio, Musumeci Olimpia, Moroni Isabella, Uziel Graziella, Santorelli Filippo M, Nesti Claudia, Filosto Massimiliano, Lamperti Costanza, Zeviani Massimo, Siciliano Gabriele

Аннотация

Myoclonus is a possible manifestation of mitochondrial disorders, and its presence is considered, in association with epilepsy and the ragged red fibers, pivotal for the syndromic diagnosis of MERRF (myoclonic epilepsy with ragged red fibers). However, its prevalence in mitochondrial diseases is not known. The aims of this study are the evaluation of the prevalence of myoclonus in a big cohort of mitochondrial patients and the clinical characterization of these subjects. Based on the database of the "Nation-wide Italian Collaborative Network of Mitochondrial Diseases," we reviewed the clinical and molecular data of mitochondrial patients with myoclonus among their clinical features. Myoclonus is a rather uncommon clinical feature of mitochondrial diseases (3.6% of 1,086 patients registered in our database). It is not strictly linked to a specific genotype or phenotype, and only 1 of 3 patients with MERRF harbors the 8344A>G mutation (frequently labeled as "the MERRF mutation"). Finally, myoclonus is not inextricably linked to epilepsy in MERRF patients, but more to cerebellar ataxia. In a myoclonic patient, evidences of mitochondrial dysfunction must be investigated, even though myoclonus is not a common sign of mitochondriopathy. Clinical, histological, and biochemical data may predict the finding of a mitochondrial or nuclear DNA mutation. Finally, this study reinforces the notion that myoclonus is not inextricably linked to epilepsy in MERRF patients, and therefore the term "myoclonic epilepsy" seems inadequate and potentially misleading.

Предметные рубрики MeSH

ИП Урсолов Владлен Сергеевич

ИНН 500118482352 · ОГРНИП 314500117700049

Реквизиты и контакты