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Journal of Child Neurology
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Effect of Topiramate on Seizures and Respiratory Dysrhythmia in Rett Syndrome

Monisha Goyal, MD

Department of Pediatrics, Division of Pediatric Neurology, University Hospitals of Cleveland and Case Western Reserve University, Cleveland, OH, monisha.goyal{at}uhhs.com

Mary Ann O'Riordan, MS

Division of Pharmacology and Critical Care, University Hospitals of Cleveland and Case Western Reserve University, Cleveland, OH

Max Wiznitzer, MD

Department of Pediatrics, Division of Pediatric Neurology, University Hospitals of Cleveland and Case Western Reserve University, Cleveland, OH

Rett syndrome, a neurodevelopmental disorder, manifests in the first few years of life with developmental arrest, stereotyped behaviors, and respiratory abnormalities. Seizures occur in 70 to 80% of patients. Clinical drug trials have not demonstrated the superiority of any specific antiepilepsy drug. We report our experience with topiramate in eight patients with Rett syndrome. Topiramate was initiated as monotherapy in two patients and as adjunctive therapy in six patients. Seven patients had improved seizure control. Respiratory abnormalities improved by 50 to 75% in two patients and by 20 to 50% in two others. In our cohort, seven of eight patients showed improvement in seizure control and/or respiratory abnormalities on topiramate. Topiramate was well tolerated. The effect of topiramate, a broad-spectrum drug, could be due to its {gamma}-aminobutyric acid (GABA)ergic and glutaminergic effects, both systems thought to be disordered in Rett syndrome. (J Child Neurol 2004;19:588-591).

Journal of Child Neurology, Vol. 19, No. 8, 588-591 (2004)
DOI: 10.1177/088307380401900804
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