Page 63 - Demo

Methylphenidate for ADHD in Smith-Magenis syndrome613Outcome measuresThe primary outcome is the change on the hyperactivity/inattention subscale of the Strengths and Difficulties Questionnaire (SDQ) during active interventional periods. Secondary outcome measures are the shortened version of the Emotion Dysregulation Inventory (EDI) reactivity index,32 Goal Attainment Scaling (GAS)33 and the personal questionnaire (PQ).34 Also, (the number of) side effects determined by the side effects checklist of MPH will be recorded.Rationale for outcome measuresThe SDQ subscale and the shortened version of the EDI have both been psychometrically considered as valid tools to measure behavior of people with ID and applicable to both children and adults.32,35,36 Specifically, the SDQ was found to be a valid outcome measure for children with ADHD symptoms and showed preliminary results of validation for children with ID.37,38 EDI was created using methods developed by the Patient-Reported Outcomes Measurement Information System (PROMIS) and validated as an efficient and sensitive method to measure emotion dysregulation in youth with ASD of any level of cognitive or verbal ability.32,36 The EDI will serve as a generalization measure that is defined as an outcome closely or more distally related to the target behavior, and is used to evaluate transfer effects of the intervention to a broader domain of functioning.39For instance, it could be the same behavior but in another setting, such as inattention at school and at home, or interventional effects on a completely different behavior, such as improved emotion regulation when the target behavior is impulsivity. In addition to the target behaviors hyperactivity and inattention in our study, measured by the SDQ, MPH might affect emotion dysregulation as well, which could be measured by the EDI. GAS is an individualized outcome measure involving goal selection and goal scaling that is standardized in order to calculate the extent to which a patient’s goals are met. Patients and/or their caregivers are allowed to choose their own specific goals in coordination with their treating physician/therapist. This makes GAS a measurement instrument that is very sensitive to change, particularly in small heterogeneous groups.Annelieke Muller sHL.indd 61 14-11-2023 09:07