MovDisordV3, Main, Exploration, bibRecord, 001D86

Coenzyme Q10‐responsive ataxia: 2‐year‐treatment follow‐up

Identifieur interne : 001D86 ( Main/Exploration ); précédent : 001D85; suivant : 001D87

Coenzyme Q10‐responsive ataxia: 2‐year‐treatment follow‐up

Auteurs : Merce Pineda [Espagne] ; Raquel Montero [Espagne] ; Asuncion Aracil [Espagne] ; Mar M. O'Callaghan [Espagne] ; Ana Mas [Espagne] ; Carmen Espinos [Espagne] ; Dolores Martinez-Rubio [Espagne] ; Francesc Palau [Espagne] ; Placido Navas [Espagne] ; Paz Briones [Espagne] ; Rafael Artuch [Espagne]

Source :

Movement Disorders [ 0885-3185 ] ; 2010-07-15.

RBID : ISTEX:4756CEB4C64F1EFAC2ADBC14ECA3E66FCADC6213

English descriptors

KwdEn :
- Adolescent, Adult, Ataxia (drug therapy), Ataxia (metabolism), Child, Child, Preschool, Female, Humans, Longitudinal Studies, Male, Neurologic Examination (methods), Statistics, Nonparametric, Time Factors, Treatment Outcome, Ubiquinone (analogs & derivatives), Ubiquinone (deficiency), Ubiquinone (therapeutic use), Vitamins (therapeutic use), Young Adult, ataxia, cerebellum, coenzyme Q10 deficiency, mitochondrial disorders, pediatric patients.
MESH :
- chemical , analogs & derivatives : Ubiquinone.
- chemical , deficiency : Ubiquinone.
- drug therapy : Ataxia.
- metabolism : Ataxia.
- methods : Neurologic Examination.
- chemical , therapeutic use : Ubiquinone, Vitamins.
- Adolescent, Adult, Child, Child, Preschool, Female, Humans, Longitudinal Studies, Male, Statistics, Nonparametric, Time Factors, Treatment Outcome, Young Adult.

Abstract

We assessed the clinical outcome after coenzyme Q10 (CoQ10) therapy in 14 patients presenting ataxia classified into two groups according to CoQ10 values in muscle (deficient or not). We performed an open‐label prospective study: patients were evaluated clinically (international cooperative ataxia rating scale [ICARS] scale, MRI, and videotape registration) at baseline and every 6 months during a period of 2 years after CoQ10 treatment (30 mg/kg/day). Patients with CoQ10 deficiency showed a statistically significant reduction of ICARS scores (Wilcoxon test: P = 0.018) after 2 years of CoQ10 treatment when compared with baseline conditions. In patients without CoQ10 deficiency, no statistically significant differences were observed in total ICARS scores after therapy, although 1 patient from this group showed a remarkable clinical amelioration. Biochemical diagnosis of CoQ10 deficiency was a useful tool for the selection of patients who are good candidates for treatment as all of them responded to therapy. However, the remarkable clinical response in 1 case without CoQ10 deficiency highlights the importance of treatment trials for identification of patients with CoQ10‐responsive ataxia. © 2010 Movement Disorder Society

Url:

https://api.istex.fr/document/4756CEB4C64F1EFAC2ADBC14ECA3E66FCADC6213/fulltext/pdf

DOI: 10.1002/mds.23129

Affiliations:

Le document en format XML

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<author><name sortKey="Espinos, Carmen" sort="Espinos, Carmen" uniqKey="Espinos C" first="Carmen" last="Espinos">Carmen Espinos</name>
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<author><name sortKey="Martinez Ubio, Dolores" sort="Martinez Ubio, Dolores" uniqKey="Martinez Ubio D" first="Dolores" last="Martinez-Rubio">Dolores Martinez-Rubio</name>
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<author><name sortKey="Navas, Placido" sort="Navas, Placido" uniqKey="Navas P" first="Placido" last="Navas">Placido Navas</name>
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<author><name sortKey="Briones, Paz" sort="Briones, Paz" uniqKey="Briones P" first="Paz" last="Briones">Paz Briones</name>
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<series><title level="j">Movement Disorders</title>
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<profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Adolescent</term>
<term>Adult</term>
<term>Ataxia (drug therapy)</term>
<term>Ataxia (metabolism)</term>
<term>Child</term>
<term>Child, Preschool</term>
<term>Female</term>
<term>Humans</term>
<term>Longitudinal Studies</term>
<term>Male</term>
<term>Neurologic Examination (methods)</term>
<term>Statistics, Nonparametric</term>
<term>Time Factors</term>
<term>Treatment Outcome</term>
<term>Ubiquinone (analogs & derivatives)</term>
<term>Ubiquinone (deficiency)</term>
<term>Ubiquinone (therapeutic use)</term>
<term>Vitamins (therapeutic use)</term>
<term>Young Adult</term>
<term>ataxia</term>
<term>cerebellum</term>
<term>coenzyme Q10 deficiency</term>
<term>mitochondrial disorders</term>
<term>pediatric patients</term>
</keywords>
<keywords scheme="MESH" type="chemical" qualifier="analogs & derivatives" xml:lang="en"><term>Ubiquinone</term>
</keywords>
<keywords scheme="MESH" type="chemical" qualifier="deficiency" xml:lang="en"><term>Ubiquinone</term>
</keywords>
<keywords scheme="MESH" qualifier="drug therapy" xml:lang="en"><term>Ataxia</term>
</keywords>
<keywords scheme="MESH" qualifier="metabolism" xml:lang="en"><term>Ataxia</term>
</keywords>
<keywords scheme="MESH" qualifier="methods" xml:lang="en"><term>Neurologic Examination</term>
</keywords>
<keywords scheme="MESH" type="chemical" qualifier="therapeutic use" xml:lang="en"><term>Ubiquinone</term>
<term>Vitamins</term>
</keywords>
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<term>Adult</term>
<term>Child</term>
<term>Child, Preschool</term>
<term>Female</term>
<term>Humans</term>
<term>Longitudinal Studies</term>
<term>Male</term>
<term>Statistics, Nonparametric</term>
<term>Time Factors</term>
<term>Treatment Outcome</term>
<term>Young Adult</term>
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<front><div type="abstract" xml:lang="en">We assessed the clinical outcome after coenzyme Q10 (CoQ10) therapy in 14 patients presenting ataxia classified into two groups according to CoQ10 values in muscle (deficient or not). We performed an open‐label prospective study: patients were evaluated clinically (international cooperative ataxia rating scale [ICARS] scale, MRI, and videotape registration) at baseline and every 6 months during a period of 2 years after CoQ10 treatment (30 mg/kg/day). Patients with CoQ10 deficiency showed a statistically significant reduction of ICARS scores (Wilcoxon test: P = 0.018) after 2 years of CoQ10 treatment when compared with baseline conditions. In patients without CoQ10 deficiency, no statistically significant differences were observed in total ICARS scores after therapy, although 1 patient from this group showed a remarkable clinical amelioration. Biochemical diagnosis of CoQ10 deficiency was a useful tool for the selection of patients who are good candidates for treatment as all of them responded to therapy. However, the remarkable clinical response in 1 case without CoQ10 deficiency highlights the importance of treatment trials for identification of patients with CoQ10‐responsive ataxia. © 2010 Movement Disorder Society</div>
</front>
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<name sortKey="Aracil, Asuncion" sort="Aracil, Asuncion" uniqKey="Aracil A" first="Asuncion" last="Aracil">Asuncion Aracil</name>
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<name sortKey="Artuch, Rafael" sort="Artuch, Rafael" uniqKey="Artuch R" first="Rafael" last="Artuch">Rafael Artuch</name>
<name sortKey="Artuch, Rafael" sort="Artuch, Rafael" uniqKey="Artuch R" first="Rafael" last="Artuch">Rafael Artuch</name>
<name sortKey="Briones, Paz" sort="Briones, Paz" uniqKey="Briones P" first="Paz" last="Briones">Paz Briones</name>
<name sortKey="Briones, Paz" sort="Briones, Paz" uniqKey="Briones P" first="Paz" last="Briones">Paz Briones</name>
<name sortKey="Espinos, Carmen" sort="Espinos, Carmen" uniqKey="Espinos C" first="Carmen" last="Espinos">Carmen Espinos</name>
<name sortKey="Martinez Ubio, Dolores" sort="Martinez Ubio, Dolores" uniqKey="Martinez Ubio D" first="Dolores" last="Martinez-Rubio">Dolores Martinez-Rubio</name>
<name sortKey="Martinez Ubio, Dolores" sort="Martinez Ubio, Dolores" uniqKey="Martinez Ubio D" first="Dolores" last="Martinez-Rubio">Dolores Martinez-Rubio</name>
<name sortKey="Mas, Ana" sort="Mas, Ana" uniqKey="Mas A" first="Ana" last="Mas">Ana Mas</name>
<name sortKey="Montero, Raquel" sort="Montero, Raquel" uniqKey="Montero R" first="Raquel" last="Montero">Raquel Montero</name>
<name sortKey="Montero, Raquel" sort="Montero, Raquel" uniqKey="Montero R" first="Raquel" last="Montero">Raquel Montero</name>
<name sortKey="Navas, Placido" sort="Navas, Placido" uniqKey="Navas P" first="Placido" last="Navas">Placido Navas</name>
<name sortKey="Navas, Placido" sort="Navas, Placido" uniqKey="Navas P" first="Placido" last="Navas">Placido Navas</name>
<name sortKey="O Callaghan, Mar M" sort="O Callaghan, Mar M" uniqKey="O Callaghan M" first="Mar M." last="O'Callaghan">Mar M. O'Callaghan</name>
<name sortKey="O Callaghan, Mar M" sort="O Callaghan, Mar M" uniqKey="O Callaghan M" first="Mar M." last="O'Callaghan">Mar M. O'Callaghan</name>
<name sortKey="Palau, Francesc" sort="Palau, Francesc" uniqKey="Palau F" first="Francesc" last="Palau">Francesc Palau</name>
<name sortKey="Palau, Francesc" sort="Palau, Francesc" uniqKey="Palau F" first="Francesc" last="Palau">Francesc Palau</name>
<name sortKey="Pineda, Merce" sort="Pineda, Merce" uniqKey="Pineda M" first="Merce" last="Pineda">Merce Pineda</name>
</country>
</tree>
</affiliations>
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Movement Disorders (revue)

Coenzyme Q10‐responsive ataxia: 2‐year‐treatment follow‐up

Coenzyme Q10‐responsive ataxia: 2‐year‐treatment follow‐up

Source :

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Abstract

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	Movement Disorders (revue)
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