Effects of creatine supplementation in Rett syndrome: a randomized, placebo-controlled trial
Freilinger M, Dunkler D, Lanator I, Item CB, Mühl A, Fowler B, Bodamer OA
Journal of developmental and behavioral pediatrics : JDBP · 16 citas
Etiquetas de revisión
Financiación no declaradaDatos neutrales que nuestra revisión registró sobre cómo se hizo este estudio. Describen el método, nunca si nos gusta el resultado.
Cómo se estudió
- Diseño
- Ensayo controlado aleatorizado (indexado por PubMed)
- Estudiado en
- Personas
- Resultado principal
- Marcadores de salud y funcionamiento
Quién lo pagó
- Financiación
- Financiación no declarada
Publicación
- Publicado
- 2011-06-08 · J Dev Behav Pediatr · vol. 32 · número 6 · págs. 454–460
- Editorial
- Lippincott Williams & Wilkins
- Citado
- 25 citas · más que el 80% de estudios similares · 1.3× el promedio de su campo
- Referencias
- 37 trabajos
- Acceso
- De pago
- Áreas de investigación
- Genetics and Neurodevelopmental Disorders · Child Nutrition and Feeding Issues · Metabolism and Genetic Disorders
- Palabras clave
- Rett syndrome, creatine monohydrate supplementation, global DNA methylation, MECP2 mutations, methionine metabolism, homocysteine remethylation, clinical symptom score, female patients
- MeSH
- humans, rett syndrome, creatine, severity of illness index, cross-over studies, double-blind method, dna methylation, dietary supplements, adolescent, adult, child, child, preschool, female, methyl-cpg-binding protein 2, tandem mass spectrometry, young adult
7 autores
- Michael Freilinger🇦🇹 · autor de correspondenciaMedical University of Vienna
- Daniela Dunkler🇦🇹Medical University of Vienna
- Ines Lanator🇦🇹 · autor de correspondenciaMedical University of Vienna
- Chike Bellarmine Item🇦🇹 · autor de correspondenciaMedical University of Vienna
- Adolf Mühl🇦🇹 · autor de correspondenciaMedical University of Vienna
- Brian Fowler🇨🇭University Children’s Hospital Basel
- Olaf A. Bodamer🇦🇹 · autor de correspondenciaParacelsus Medical University; Medical University of Vienna
Resumen
El texto original del estudio, en el idioma en que se publicó.
Objective
To evaluate the effects of creatine monohydrate (CMH) supplementation on global DNA methylation and disease-specific clinical symptoms in female patients with Rett syndrome (RTT).
Methods
Double-blind, randomized, placebo-controlled crossover trial of female patients with RTT. Participants received 200 mg/kg of either CMH or placebo daily for 6 months and switched following a 4-week washout period. Primary endpoints were change in global DNA methylation and in a RTT-specific symptom score as defined by medical history and clinical evaluation with Rett Syndrome Motor and Behavioral Assessment. Secondary endpoints were changes in biochemical markers of methionine metabolism.
Results
Eighteen female patients aged 3 to 25 years with clinically diagnosed typical RTT and MECP2 mutation at clinical Stages III or IV were studied. CMH supplementation resulted in a statistically significant increase of global methylation by 0.11 (95% confidence interval 0.03-0.19, p = .009) compared with placebo. Total and subscores of Rett Syndrome Motor and Behavioral Assessment tended to improve but without statistical significance.
Conclusion
CMH supplementation increases global DNA methylation statistically significantly. Scores were lower for creatine than for placebo reflecting clinical improvement but not reaching statistical significance. Biochemical variables of methionine-homocysteine remethylation are unaffected. Multicenter studies are urgently warranted to evaluate the long-term effects of CMH supplementation in an optimally homogenous RTT population over a prolonged period.
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