The Duchenne connect experience with a pain interference patient reported outcome measure
Chronic pain can be a significant problem for many with neuromuscular disorders, including Duchenne and Becker muscular dystrophy (DBMD). Pain, as a subjective experience, is intended to be self-reported, and a wide range of pain patient-report outcome measures (PROs) exist. A review of the literature found a single study that focused specifically on pain in children with DBMD (N = 53) and one of adults (N = 80); this suggests an unmet need. DuchenneConnect is an online, self-report registry. In February 2016 DC integrated the pediatric and adult 6-item PROMISTM Item Banks for Pain Interference to explore the impact of pain in DBMD. The incorporation of the pediatric pain interference measure represents the first use of a pediatric PRO in the registry's history. Thus we included a question to assess who responded. From February 5 to March 22, 2016 we obtained 148 pediatric and 52 adult surveys. On preliminary results, adults reported a mean pain score of 2.0 (SD 1.3), which is below the midpoint on a scale of 1–5. 13.5% (n = 7) of adults reported a mean of >4. Children reported a mean of 2.2 (SD 1.0) and 3.4% (n = 5) reported a mean of >4. There was no significant correlation between pain interference and age. Preliminary results show relatively low pain interference in a registry population, though 6% report high pain. While the survey instructs self-report of data, 85.8% of caregivers provided answers without input from the child and 40.4% provided answers without input from the adult participant. Given that pain experience is subjective, self-report is vital. These results demonstrate a limitation that may be prevalent in self-report research, and one identified only due to a non-standard practice of adding a reporting item to the measure. Regular integration of a question about who is providing answers to PRO measures will improve their integration into self-report registries, allow for data quality checks, and indicate when additional education is needed.