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INTRODUCTION: Health-state utilities (HSUs) summarise health-related quality of life for economic evaluation. Estimates in haemophilia vary by population, treatment context, instrument, value set, and recruitment setting. AIM: To synthesise HSU estimates from multi-attribute utility instruments in adults with haemophilia and evaluate the interpretability of a single pooled value. METHODS: Six databases were searched from inception to 30 June 2026. The primary meta-analysis included one patient-reported EQ-5D or SF-6D mean, with usable SD, from each independent adult cohort. A REML random-effects model used Hartung-Knapp confidence intervals. Prediction intervals, leave-one-out analysis, alternative estimators, eligibility and overlap scenarios, and Egger regression assessed robustness. Leave-one-out estimates ranged from 0.668 to 0.690. Egger's intercept was -6.24 (p = 0.003), indicating asymmetry rather than proving publication bias. Data were insufficient for robust subgroup meta-analysis. CONCLUSION: Adults with haemophilia experience a substantial HRQoL burden; however, variability in pooled HSU estimates means that this value should be interpreted as a broad reference point rather than a precise estimate for every setting. Future research would benefit from clearer adult eligibility criteria, more consistent reporting of variance, and broader geographic and population representation, including women and girls with haemophilia and symptomatic carriers. CILINICAL TRIAL REGISTRATION INFORMATION: This systematic review was prospectively registered in PROSPERO (CRD420251234536). Clinical trial registration is not applicable.
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Zhang et al. (2026) studied this question.
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