Glycopyrrolate and Theophylline for the Treatment of Severe Pallid Breath-holding Spells

Pediatric Cardiology, Department of Pediatrics, University of Parma, Italy.
PEDIATRICS (Impact Factor: 5.47). 03/2013; 131(4). DOI: 10.1542/peds.2012-0182
Source: PubMed


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.

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Available from: Ilaria Bo, Jan 09, 2016
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    • "This drug is approved for treatment of myoclonus but not FDA approved for breath holding spells. Glycopyrrolate, theophylline, fluoxetine, and levetiaciteram have been used to treat breath holding spells in individual cases and need further studies to confirm efficacy and safety [31–33]. "
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    ABSTRACT: Breath holding spells are a common and dramatic form of syncope and anoxic seizure in infancy. They are usually triggered by an emotional stimuli or minor trauma. Based on the color change, they are classified into 3 types, cyanotic, pallid, and mixed. Pallid breath holding spells result from exaggerated, vagally-mediated cardiac inhibition, whereas the more common, cyanotic breathholding spells are of more complex pathogenesis which is not completely understood. A detailed and accurate history is the mainstay of diagnosis. An EKG should be strongly considered to rule out long QT syndrome. Spontaneous resolution of breath-holding spells is usually seen, without any adverse developmental and intellectual sequelae. Rare cases of status epilepticus, prolonged asystole, and sudden death have been reported. Reassurance and education is the mainstay of therapy. Occasionally, pharmacologic intervention with iron, piracetam; atropine may be of benefit. Here we present 2 cases, one of each, pallid and cyanotic breath holding spells.
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    ABSTRACT: Cyanotic breath-holding spells are generally benign and resolve spontaneously by 4 to 5 years of age. Treatment with iron and other drugs has been employed in selected cases with very frequent and severe episodes. We describe a 10-year-old boy with recent-onset cyanotic breath-holding spells that were activity limiting. He was unable to participate in physical activities with his peers as any argument or emotional upset provoked these spells. Treatment with oral iron and piracetam was ineffective. However, treatment with oral theophylline produced dramatic amelioration of symptoms, and he was once again able to participate in play activities with his peers. We believe that general central nervous system stimulant and respirogenic effects of theophylline were instrumental in control of symptoms in our child.
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