Developmental Medicine & Child Neurology · 2007 · 43 citations · 21 references
MedicineNeurosciencePediatricsNeurochemistryNeurodegenerationPediatric NeurologyCerebrospinal Fluid BiomarkersDevelopmental NeurologyInfant Neurological DevelopmentNeonatal Metabolic DisordersNeurodevelopmental DisordersPediatric Neurologic DisordersCerebrospinal Fluid SystemMetabolic EncephalopathyNeonatal Brain MetabolismNeuroimaging Of Metabolic DisordersNeurotransmitter PhenotypingNeurotransmitter System Alterations
Neurotransmitters are essential in young children for differentiation and neuronal growth of the developing nervous system. We aimed to identify possible factors related to secondary neurotransmitter abnormalities in pediatric patients with neurological disorders. We analyzed cerebrospinal fluid (CSF) and biogenic amine metabolites in 56 infants (33 males, 23 females; mean age 5.8mo [SD 4.1mo] range 1d-1y) with neurological disorders whose aetiology was initially unknown. Patients were classified into three clinical phenotypes: epileptic encephalopathy, severe motor impairment, and non-specific manifestations. All patients showed normal results for screening of inborn errors of metabolism. We report clinical, neuroimaging, and follow-up data. Among the patients studied, 10 had low homovanillic acid (HVA) levels and in four patients, 5-hydroxyindoleacetic acid (5-HIAA) was also reduced. Patients with neonatal onset had significantly lower levels of HVA than a comparison group. HVA deficiency was also associated with severe motor impairment and the final diagnosis related to neurodegenerative disorders. 5-HIAA values tended to be decreased in patients with brain cortical atrophy. The possibility of treating patients with L-Dopa and 5-hydroxytryptophan, in order to improve their neurological function and maturation, may be considered.
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The metabolic and molecular bases of inherited disease
Ying-Tsong Chen · Medical Entomology and Zoology · 2001 · 6.9K citations
Luisa Bonafé, Beat Thöny, Johann Penzien et al. · The American Journal of Human Genetics · 2001 · 237 citations · Full text
Rapid‐onset dystonia‐parkinsonism
William B. Dobyns, Laurie J. Ozelius, P. L. Kramer et al. · Neurology · 1993 · 191 citations
Life Sciences, Genetics, Neurology +16