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Pallid Breath-holding Spells

Evaluation of the Autonomic Nervous System

Division of Neurology, The Children's Hospital of Philadelphia and the Departments of Neurology and Pediatrics, University of Pennsylvania, Philadelphia, Pennsylvania

Claire M. Chee, RN

Division of Neurology, The Children's Hospital of Philadelphia and the Departments of Neurology and Pediatrics, University of Pennsylvania, Philadelphia, Pennsylvania

Peter H. Berman, MD

Division of Neurology, The Children's Hospital of Philadelphia and the Departments of Neurology and Pediatrics, University of Pennsylvania, Philadelphia, Pennsylvania

Excessive vagal tone has been implicated as the cause of pallid breath-holding spells (PBHS) in children. The following study was undertaken to test the hypothesis that children with PBHS have underlying autonomic nervous system (ANS) dysfunction. Five patients (age 29-79 months old) who had experienced PBHS were evaluated at a time when they were clear of spells for ANS dysfunction. A battery of clinical bedside tests were given. The results were compared with test results of a control group of children (age 18-104 months) who had not experienced PBHS. Data collected included blood pressure, pulse rate, mean arterial pressure, the 30:15 R-R ratio upon standing, the expiratory:inspiratory (E:I) R-R ratio, pupillary response to conjunctival pilocarpine instillation, and plasma norepinephrine levels upon standing.

Breath-holders displayed a statistically significant (p < 0.05) percent decrease in mean arterial pressure (-10.2% PBHS vs. -4.1% controls) and an unsustained increase in pulse rate during the lying to standing maneuver. Two children with PBHS had "positive orthostatic signs," and one child with PBHS had a plasma norepinephrine level of 94 pg/ml (60% below the mean for both groups). There is evidence to suggest a subtle, underlying, generalized autonomic dysfunction in children with PBHS.

A strong familial tendency toward syncope, breath-holding spells, and seizures was recognized in nine of ten subjects. Additionally, there was a strong influence from the maternal side of the family in seven of nine subjects.

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