The abnormalities of dopaminergic activity in the basal ganglia have been emphasized to be effective in dystonia. We hereby report a case of a 2.5-year-old male patient who presented with tonic-clonic sezures and who developed dystonia after being given phenytoin. Biperidene hydrochloride was administered intramuscularly; primidone was added to the treatment regimen. After a 7-day-follow-up at the hospital, the patient had no dystonia and was discharged.Key words: epilepsy, phenytoin, dystonia.Although the underlying neuro-chemical pathology is not known, it has been emphasized that abnormalities seen at dopaminergic activity in the basal ganglions are effective in dystonia. It is mostly the side effect of classical antipsychotics; it may also emerge after taking medicines such as metoclopramide, which are effective on the dopaminergic pathway. 1 The acutely starting extrapyramidal symptoms in childhood are accepted as an urgent medical condition and they need to be alleviated as soon as possible.Phenytoin, which is a derivative of hydantoin, is widely used in the treatment of tonic-clonic seizure due to its anticonvulsant activity without a sedative effect. In our case, acute dystonia developed approximately 12 hours following phenytoin infusion.
Case ReportA 2.5-year-old-male patient presented to the hospital with convulsions lasting for approximately 5 minutes and repeating 4-5 times in half an hour. The patient, born through spontaneous vaginal delivery (2,900 g), had normal development stages. His 11-year-old brother was suffering from epilepsy, diagnosed at the age of four; and was on sodium valproate therapy. Since the patient had an epileptic attack 2 months ago, brain MR was taken to exclude intracranial pathology. Brain MRI of the patient had demonstrated a slight increase at the centrum semioviale level and bilateral forceps major levels in the white matter on T2-weighed images and flair images. It was considered that myelinization had not completed its development in the white matter. As a result of detecting bilateral generalized sharp wave paroxysm in his electroencephalography (EEG), sodium valproate 20 mg was begun.After 1-month of sodium valproate-treatment, the patient re-presented to the emergency department due to generalized tonic-clonic seizures. Rectal diazepam (10 mg) was administered, which was followed by phenytoin (20 mg/kg). The patient had status epilepticus, and was transferred to the intensive care unit and midazolam infusion was started. Afterwards, the dose of midazolam was tapered gradually as his seizures subsided; an additional dose of phenytoin (10 mg/kg) was administered.His physical examination revealed: temperature 36.7ºC, blood pressure 95/55 mm Hg, pulse rate 95/min., oxygen saturation 98%; frontal fontanelle was closed, light reaction was positive on both sides with equal sized pupils, and no neck stiffness and pathological reflex. Deep tendon reflexes were normal. Muscle strength was 5/5 for all extremities. Laboratory examination revealed: glucose 90 mg/dl, urea 33 mg...