Currently, data regarding the burden of Huntington’s disease (HD) are scarce, and no specific self-reporting assessment instruments are available. There is a clear need for the design of self-reporting techniques that address patient reported outcomes (PRO), caregiver burden (CGB), and caregivers’ healthcare resource utilization (RU) in HD.
We developed the Huntington Self-Assessment Instrument (HSAI), a tool that assesses PRO, CGB, and RU specifically in HD. The HSAI is split into two sections: the Huntington Self-Reported Instrument (H-SRI) and the Huntington Resources Utilization Interview (H-RUI). Each of the sections contain a questionnaire; one designed for the patient and the other for the caregiver. The patient section of the H-SRI is based on the Unified HD Rating Scale (UHDRS), with three parts that assess motor, behavioral, and functional capacity. It was critical to establish a self-reported assessment that would be a good proxy for the UHDRS clinical motor evaluation, an evaluation normally performed by an experienced practitioner. Visual analogue scales were therefore used to optimize patients’ understanding of the questionnaire, and maximize assessment accuracy. The caregiver part of the H-SRI consists largely of the validated HD Quality of Life Battery for Carers (HDQoL-C). The patient part of the H-RUI assesses direct and indirect healthcare RU; the caregiver part determines the out-of-pocket costs and the opportunity costs of patient care.
The HSAI will be available in English, German, French, Spanish, Italian, and Swedish; the Swedish version will be validated first. The UHDRS, Hospital Anxiety and Depression Scale (HAD), EQ-5D, SF-36, and the Zarit scale will be used for external validation of the HSAI. Internal validation will be carried out using standard methods. HSAI validation data will be presented at the meeting.
The HSAI is an important development that may be used to generate data required for appropriate policy on HD management.
P oster 22: The Huntington Self-Assessment Instrument (HSAI): A Comprehensive and Specific Tool to Measure the Burden of Huntington’s Disease. Available from: https://www.researchgate.net/publication/247343946_P_oster_22_The_Huntington_Self-Assessment_Instrument_%28HSAI%29_A_Comprehensive_and_Specific_Tool_to_Measure_the_Burden_of_Huntington%27s_Disease [accessed Apr 2, 2015].