Pipecolic acid elevation in plasma and cerebrospinal fluid of two patients with pyridoxine‐dependent epilepsy

Abstract
Diagnosis of pyridoxine‐dependent epilepsy is based on the clinical response to high‐dosage application of pyridoxine. Here, we report on 2 patients with pyridoxine‐dependent epilepsy with significant elevation of pipecolic acid concentrations in plasma and cerebrospinal fluid (CSF) and further increase of pipecolic acid in CSF during a 72‐hour pyridoxine withdrawal in 1 of them. Patients with non–pyridoxine‐dependent epilepsy had normal pipecolic acid concentrations in plasma and significantly lower concentrations in CSF. High plasma and CSF pipecolic acid concentrations might provide a diagnostic marker in pyridoxine‐dependent epilepsy. Ann Neurol 2000;48:121–125

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