Generic preference‐based health‐related quality of life in children with neurodevelopmental disorders: a scoping review

Abstract

AIM To describe how generic preference-based health-related quality of life (HRQoL) instruments have been used in research involving children with neurodevelopmental disorders (NDD).METHOD A systematic search of nine databases identified studies that used generic preference-based HRQoL instruments in children with NDD. Data extracted following the Preferred Reporting Items for Systematic Review and Meta-Analyses extension for Scoping Review guidelines included type of NDD, instrument used, respondent type, justification, and critical appraisal for these selections.RESULTS Thirty-six studies were identified: four cost-utility analyses; 15 HRQoL assessments; five economic burden studies; three intervention studies; and nine 'other'. The Health Utilities Index (Mark 2 and Mark 3) and EuroQoL 5D (EQ-5D; three-level EQ-5D, five-level EQ-5D, and the youth version of the EQ-5D) instruments were most frequently used (44% and 31% respectively). The relatively low use of these instruments overall may be due to a lack of psychometric evidence, inconsistency in justification for and lack of clarity on appropriate respondent type and age, and geographical challenges in applying preference weights.INTERPRETATION This study highlights the dearth of studies using generic preference-based HRQoL instruments in children with NDD. The use of cost-utility analysis in this field is limited and validation of these instruments for children with NDD is needed. The quality of data should be considered before guiding policy and care decisions.

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The Cost-effectiveness of screening tools used in the diagnosis of fetal alcohol spectrum disorder: A modeled analysis

“…Though quality adjusted life years (QALY) are commonly recommended for use as the primary outcome measure in economic evaluations [19], at present the impact of an FASD diagnosis on patients' health related quality of life (HRQoL) is not understood [20]. As a substitute to QALY, the number of diagnoses or other diagnoses based outcomes are sometimes used as a primary 6 effectiveness measure in CEA of screening strategies [21].…”