Prospective evaluation of clinical prediction rules for brief resolved unexplained event

Published clinical prediction rules propose to determine risks of event recurrence and serious underlying condition in infants with brief resolved unexplained event (BRUE). The objective of this study was to evaluate these rules in a prospectively recruited cohort of infants with BRUE to test the hypothesis that the rules may be helpful in predicting risk but their test characteristics may differ from those previously reported.

To determine the validity of prediction rules for risk of recurrent events and serious diagnoses, we applied the calculators to patients with BRUE who were prospectively recruited at our institution. Infants were recruited during their index hospitalization, caregiver-reported symptoms were obtained by questionnaires during the 6-month follow-up period, and charts reviewed for clinical data. Receiver operating characteristic (ROC) analyses were used to calculate values for the sensitivity and specificity of each calculator.

The cohort included 178 subjects and 73% (130) completed questionnaires. Overall, 78% (101/130) had persistent caregiver-reported symptoms, 12% (16/130) recurrent BRUE, and 28% (50/178) a serious condition diagnosis. The most common serious condition was oropharyngeal dysphagia with aspiration (78% of serious diagnoses, representing 22% of the cohort). On ROC analysis, area under the curve was 0.7 (95% confidence interval [CI]: 0.57-0.83, p = 0.004) for recurrent BRUE and 0.59 (95% CI: 0.49-0.68, p = 0.08) for serious underlying condition. The calculators provided 81% sensitivity, 52% specificity, 20% positive predictive value, and 96% negative predictive value for predicting recurrent BRUE and 54% sensitivity, 68% specificity, 40% positive predictive value, and 79% negative predictive value for predicting a serious condition in our cohort.

Our results suggest that published clinical prediction rules may accurately predict event recurrence but may have relatively low discriminatory power and tend to underestimate risk of serious underlying diagnoses, which may limit their clinical impact.