Successful Treatment of Pallid Breath-holding Spells With Fluoxetine

Abstract

Objective: To promote dialogue among readers and between readers and editors, we offer here brief reports and observations on topics of interest authored by members of the AAP Grand Rounds editorial team.At the severe end of the clinical spectrum, pallid breath-holding (PBH) spells can be disruptive and debilitating to the patients and families of those who are afflicted. Effective treatment options are few and include medications,1 pacemaker implantation,2 and tincture of time.3In their recent publication, Walsh and colleagues4 seek to add to our clinical armamentarium for the medical treatment of PBH spells using fluoxetine. Fluoxetine, a selective serotonin reuptake inhibitor (SSRI), has been shown to be beneficial in the treatment of neurocardiogenic syncope in older children and adults.5 Hypothesizing that PBH spells are physiologically similar to vasovagal syncope, these investigators performed a retrospective review and attempted a prospective follow-up on all patients referred to their institution for severe PBH spells treated with fluoxetine after failing “medical management.” All patients had been screened for conditions associated with cardiac syncope and sudden cardiac death. Each had documented severe bradycardia or asystole during PBH episodes.The study included 6 (out of 7) patients, age 4 to 24 months, identified by retrospective chart review who were available for prospective follow-up. Four patients had failed alternative medications (propranolol, carbamazepine, and/or glycopyrrolate) prior to initiating fluoxetine. Fluoxetine treatment was maintained for 1 to 2 years and was associated with improved symptoms based on medical records review and parental report in 5 of 6 patients (3 complete resolution, 2 partial resolution). No serious adverse events from fluoxetine therapy occurred. Side effects included decreased appetite and tremors, both of which improved over time on therapy. The authors conclude that fluoxetine is a safe and effective first-line agent for the treatment of PBH spells.Though encouraging and thought-provoking, their study by its design provides little hard evidence upon which to base a treatment recommendation. Outcome measures were subjective and the treatment was uncontrolled and unblinded. Though apparently benign in the small cohort of patients treated in this study, there is no large clinical experience with the use of SSRIs in children of the age typically affected by PBH spells. As a result, the safety of these medications cannot be readily extrapolated from this small cohort. Nonetheless, when faced with pacemaker implantation as the only other valid treatment option, the notion of prescribing treatment with fluoxetine is appealing.It is often the case that pediatric diseases are too rare and varied in nature to lend themselves well to study by randomized controlled trials. However, the prevalence of severe breath-holding spells (those associated with loss of consciousness with or without convulsions) has been estimated as high as 4.6%6 and simple breath-holding spells have a prevalence as high as 27%.7 Walsh and colleagues have recommended fluoxetine be used as first-line therapy for severe PBH spells based on preliminary evidence. It is incumbent upon those of us who care for these patients to subject fluoxetine to a proper controlled trial before heeding that advice without careful consideration.