期刊
NEUROSCIENCE AND BIOBEHAVIORAL REVIEWS
卷 33, 期 3, 页码 414-421出版社
PERGAMON-ELSEVIER SCIENCE LTD
DOI: 10.1016/j.neubiorev.2008.11.001
关键词
Phenylketonuria; Phenylalanine; Meta-analysis; Neuropsychological speed test; Treatment recommendations
资金
- German Federal Ministry of Education and Research [01GM0305]
Although pathogenesis of phenylketonuria is not completely understood, a low phenylalanine diet is effective to prevent severe neurological impairment, mental retardation and behavioural difficulties. Treatment recommendations heavily rely on neuropsychological research; however, single study results are ambiguous, what is reflected in substantial variation of US, British, German, Dutch and French recommendations for blood phenylalanine concentrations for adolescents and adults. We conducted a meta-analysis estimating the influence of age, phenylalanine level, and type of neuropsychological test on effect sizes (standardized differences between controls and patients) of computer-based speed measurements in phenylketonuric patients. The effect of blood phenylalanine level on effect size was more pronounced in children and adolescents than in adults, with choice reaction time being particularly sensitive for phenylalanine concentrations. Results corroborate all recommendations for children. With the exception of the US and Dutch recommendations, all recommendations for adolescents seem to be too liberal. The same effect size is predicted for adult phenylalanine concentrations between 750 and 1500 mu mol/L not suggesting a preference for any of the published treatment recommendations for adulthood. (c) 2008 Elsevier Ltd. All rights reserved.
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