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Outcomes and outcome measurement instruments1155The aim of this scoping review was to provide an overview of outcomes and outcome measurement instruments selected in clinical trials in individuals with ID and GNDs, measured by COAs focusing on neurological functioning, mental and social functioning, and the overarching concepts of perceived health and health-related quality of life (HR-QoL). MethodsWe followed the Preferred Reporting Items for Systematic Reviews and Meta-Analysis (PRISMA) Protocols, and the PRISMA extension for Scoping Reviews (PRISMA-ScR) checklist (See Supplementary Figure 1).32,33 The methodological framework was published in advance in the Open Science Framework: https://osf.io/2zmxv/. EligibilityClinical trials for any intervention were included in the review, including comparative studies (randomized and non-randomized), single-case trials and single-arm case series (retrospective and prospective), and trial protocols. Validation and feasibility studies, and economic evaluations were excluded. GNDs were defined as disorders with a genetic etiology affecting the nervous system in early development. GNDs associated with ID were included. ID was defined as substantial limitations in both intellectual functioning and adaptive behavior, originating during the developmental period.5 Neurometabolic disorders, consisting of a subgroup of rare genetic hereditary conditions in which the impairment of a biochemical pathway is essential to the pathophysiology of the disease,34 were included in case they are associated or presented with intellectual deficits/impairment. Studies were included when a participant showed intellectual impairments. Exclusion criteria included ID explicitly stated to be due to exogenous factors.Studies were included if these used a COA, i.e., a PRO, ClinRO, ObsRO, or PerfO (Box 1; Figure 1). Condition-specific and personalized outcome measures were included as well (Box 1). Biological and physiological variables were excluded. Clinical trials with only epilepsy characteristics or Annelieke Muller sHL.indd 115 14-11-2023 09:07