Sagittal craniosynostosis: a utility outcomes study

OBJECTIVE Sagittal craniosynostosis results in a characteristic scaphocephalic head shape that is typically corrected surgically during a child's 1st year of life. The authors' objective was to determine the potential impact of being born with sagittal craniosynostosis by using validated health state utility assessment measures. METHODS An online utility assessment was designed to generate health utility scores for scaphocephaly, monocular blindness, and binocular blindness using standardized utility assessment tools, namely the visual analog scale (VAS) and the standard gamble (SG) and time trade-off (TTO) tests. Utility scores were compared between health states using the Wilcoxon and Kruskal-Wallis tests. Univariate regression was performed using age, sex, income, and education as independent predictors of utility scores. RESULTS Over a 2-month enrollment period, 122 participants completed the online survey. One hundred eighteen participants were eligible for analysis. Participants rated scaphocephaly due to sagittal craniosynostosis with significantly higher (p < 0.001) median utility scores (VAS 0.85, IQR 0.76-0.95; SG 0.92, IQR 0.84-0.98; TTO 0.91, IQR 0.84-0.95) than both monocular blindness (VAS 0.60, IQR 0.50-0.70; SG 0.84, IQR 0.68-0.94; TTO 0.84, IQR 0.67-0.91) and binocular blindness (VAS 0.25, IQR 0.20-0.40; SG 0.51, IQR 0.18-0.79; TTO 0.55, IQR 0.36-0.76). No differences were noted in utility scores based on participant age, sex, income, or education. CONCLUSIONS Using objective health state utility scores, authors of the current study demonstrated that the preoperatively perceived burden of scaphocephaly in a child's 1st year of life is less than that of monocular blindness. These relatively high utility scores for scaphocephaly suggest that the burden of disease as perceived by the general population is low and should inform surgeons' discussions when offering morbid corrective surgery, particularly when driven by aesthetic concerns.