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Prospective evaluation of clinical prediction rules for brief resolved unexplained event
Daniel R Duncan1, Clare Golden1, Michael Kim1
1Aerodigestive Center, Division of Gastroenterology, Hepatology and Nutrition, Boston Children's Hospital, Boston, Massachusetts, USA.
Insights
Clinical prediction rules for brief resolved unexplained event (BRUE) may predict recurrence but underestimate serious conditions. These tools may have limited clinical impact due to lower discriminatory power.
Area of Science:
- Pediatrics
- Clinical Prediction Models
- Infant Health
Background:
- Brief Resolved Unexplained Event (BRUE) requires accurate risk assessment for recurrence and serious underlying conditions.
- Existing clinical prediction rules aim to stratify these risks, but their real-world performance needs validation.
Purpose of the Study:
- To prospectively evaluate the accuracy and test characteristics of published clinical prediction rules for infants with BRUE.
- To test if these rules accurately predict event recurrence and serious diagnoses in a new cohort.
Main Methods:
- Prospective recruitment of infants with BRUE during hospitalization.
- Caregiver questionnaires for symptom reporting over 6-month follow-up.
- Chart review for clinical data and ROC analysis for rule validation.
Main Results:
- The cohort included 178 infants; 28% had serious conditions, most commonly oropharyngeal dysphagia with aspiration.
- Rules showed 81% sensitivity and 52% specificity for recurrent BRUE.
- For serious conditions, rules had 54% sensitivity and 68% specificity, with a low positive predictive value (40%).
Conclusions:
- Published prediction rules may accurately identify recurrent BRUE but possess limited discriminatory power.
- These rules tend to underestimate the risk of serious underlying diagnoses in infants with BRUE.
- The findings suggest a potential limitation in the clinical utility of current prediction rules for BRUE management.
Objectives:
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.
Methods:
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.
Results:
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.
Conclusions:
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.
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