Dr. Gliksman of Hackensack University Medical Center and Hackensack Meridian Health School of Medicine at Seton Hall University has no relevant financial relationships to disclose.)
Breath-holding spells, described more than 400 years ago, are paroxysmal clinical events that occur between the ages of 6 months and 4 to 6 years in which vigorous crying is interrupted by end-expiratory apnea, followed by cyanosis or pallor, loss of consciousness, and occasionally by a clonic seizure or myoclonic movements. Though virtually always triggered by a stimulus (pain, fear, or anger), the misconception still exists that the child “does it on purpose.” They are terrifying to parents or caregivers, but are often dismissed by clinicians in a cavalier manner due to their benign long-term outcome and the misconception that they occur in “spoiled children.” This article reflects studies regarding presumed autonomic dysregulation, rare occurrences of asystole, and seizures. The authors expand on recent studies proposing possible treatment options in children with breath-holding spells.
Historical note and terminology
Breath-holding spells are paroxysmal clinical events in infants and young children in which vigorous crying is typically followed by a relatively brief period of apnea, cyanosis or pallor, loss of consciousness, and occasionally, by clonic movements of the limbs. The term is a perhaps a misnomer. “Breath-holding spells” suggests a volitional aspect in the suspension of breathing following inspiration, whereas these events are typically are associated with end-expiratory apnea and cannot be completely reproduced voluntarily by the child.
References to breath-holding spells appear in the literature as early as the writings of Nicholas Culpeper (1616-1654): "There is a disease. . . in children from anger or grief, when the spirits are much stirred and run from the heart to the diaphragms forceably, and hinder or stop the breath. . . but when the passion ceaseth, this symptom ceaseth" (Gauk et al 1963).
Breath-holding spells were later characterized as paroxysmal but nonepileptic events (Maulsby and Kellaway 1964; Lombroso and Lerman 1967). Early investigators also brought to attention the differentiation between cyanotic and pallid breath-holding spells. Since that time, there have been numerous hypotheses and theories regarding the etiology, prognosis, and management of breath-holding spells. Various terms have been used to describe the less common pallid breath-holding spells (including reflex anoxic seizures, white breath-holding attacks, pallid infantile syncope, and nonepileptic vagal attacks). In contrast, the cyanotic type are usually implied by the term breath-holding spells or referred to as cyanotic infantile syncope.
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