Compulsive respiratory stereotypies in children with autistic features: polygraphic recording and treatment with fenfluramine.
Syncopes due to compulsive respiratory stereotypies were studied in eight patients with autistic features. Most had been referred for episodes thought to be intractable epileptic seizures. Polygraphic recording showed two types of syncope, one induced by prolonged apnea and the other by a prolonged Valsalva maneuver. Fenfluramine, 1.5-3 mg/kg per day, was given in an open trial. In four of five cases with frequent Valsalva maneuvers, respiratory stereotypies and syncopes were suppressed for 2-18 months. Patients with periodic apneas were more severely retarded and had less clear benefit. Side effects consisted of dose-dependent sedation and mild weight loss which stabilized without interrupting treatment. We suggest that these syncopes are volitional and may be associated with pleasant sensations. A double-blind placebo-controlled trial of fenfluramine seems warranted in such patients.
Gastaut
H
H
Institut de Recherches Neurologiques, Faculte de Medecine, Marseille, France.
Zifkin
B
B
Rufo
M
M
D001049:Apnea / Q000209:etiology; D001321:Autistic Disorder / Q000150:complications / Q000175:diagnosis / Q000188:drug therapy*; D002648:Child; D002675:Child, Preschool; D003192:Compulsive Behavior / Q000188:drug therapy* / Q000209:etiology; D004562:Electrocardiography; D004569:Electroencephalography; D005260:Female; D005277:Fenfluramine / Q000009:adverse effects / Q000627:therapeutic use*; D006801:Humans; D008297:Male; D013239:Stereotyped Behavior / Q000188:drug therapy* / Q000209:etiology; D013575:Syncope / Q000188:drug therapy* / Q000209:etiology; D014636:Valsalva Maneuver; D014836:Volition
D005277:Fenfluramine