title: Glycopyrrolate and theophylline for the treatment of severe pallid breath-holding spells.
abstract: Severe pallid breath-holding spells (BHSs) are based on parasympathetic hyperactivity, leading to cardiac asystole, pallor, brain ischemia, loss of consciousness, and reflex anoxic seizures. In recent years, an increasing number of patients with severe pallid BHSs have been successfully treated with pacemaker implantation. We present the case of a 13-month-old girl suffering from repeated severe pallid BHSs, causing asystole, loss of consciousness, and generalized anoxic seizures. She underwent treatment with oral glycopyrrolate, an anticholinergic drug, and an oral retard preparation of theophylline. The aim of the treatment was to decrease cardiac inhibition with glycopyrrolate and to bring about a positive chronotropic effect with theophylline. In our case, the combined therapy was effective in suppressing syncope and reflex anoxic seizures associated with BHSs This avoided the need for ventricular pacemaker implantation.
authors.affiliation: Pediatric Cardiology, Department of Pediatrics, University of Parma, Via Gramsci 14, 43126 Parma, Italy. nicola.carano@unipr.it
authors.forename: Nicola
authors.initials: N
authors.lastname: Carano
authors.forename: Ilaria
authors.initials: I
authors.lastname: Bo
authors.forename: Elena
authors.initials: E
authors.lastname: Zanetti
authors.forename: Bertrand
authors.initials: B
authors.lastname: Tchana
authors.forename: Giulia
authors.initials: G
authors.lastname: Barbato
authors.forename: Aldo
authors.initials: A
authors.lastname: Agnetti
mesh_terms: D001049:Apnea / Q000150:complications / Q000175:diagnosis / Q000188:drug therapy*; D062485:Breath Holding; D005260:Female; D006024:Glycopyrrolate / Q000627:therapeutic use*; D006801:Humans; D007223:Infant; D018727:Muscarinic Antagonists / Q000627:therapeutic use*; D010726:Phosphodiesterase Inhibitors / Q000627:therapeutic use*; D012008:Recurrence; D012640:Seizures / Q000188:drug therapy / Q000209:etiology; D013575:Syncope / Q000188:drug therapy / Q000209:etiology; D013806:Theophylline / Q000627:therapeutic use*
chemical_list: D018727:Muscarinic Antagonists; D010726:Phosphodiesterase Inhibitors; D013806:Theophylline; D006024:Glycopyrrolate