Abstract

Abstract Background/Aims The Stickler syndrome is associated with functional changes in one of the COL2 (type 1, common), COL11 (type 2, uncommon) or COL9 (very rare) genes and causes early onset osteoarthritis. Previous studies have looked at the factors associated with response to pain management interventions in osteoarthritis, but none have done so in a Stickler syndrome cohort. We aimed to identify the factors associated with response to a pain management clinic in patients with osteoarthritis caused by Stickler syndrome. Methods All attendees completed the Brief Pain Inventory (BPI). Scores from 321 Stickler syndrome patients were gathered during each attendance of clinic. Repeat attenders were categorised into responders (BPI score improved on average by > 2 points) and non-responders (BPI score worsened on average by > 2 points). The responders (R) and non-responders (NR) cohorts were characterised and compared using variables including initial pain scores, pain management used, pain categories affected, time between visits, sex, age, genetic mutations, pain location and presence of joint replacements. Results 237/321 patients re-attended the clinic. 26/237 were Responders (R) and 30/237 were Non-responders (NR). All baseline BPI scores were statistically significantly higher in the R cohort, suggesting that the clinic provides greater benefit to those with greater levels of pain. Patients in the total cohort who started with high pain scores (7 or above) had an improvement in scores compared to those who started with low pain scores (3 or below), who had a worsening of scores (-1.158 vs 0.8194, p < 0.0001). Furthermore, there was a weak negative correlation between initial pain score and change in score (r2=0.136). Topical NSAIDs were more commonly used in the NR cohort (33.33% in NR vs 7.69% in R, p = 0.0248) whereas oral NSAIDs were more commonly used in the R cohort (6.67% in NR vs 30.77% in R, p = 0.0332). A final difference between the cohorts was the effect of the pain on the different interference scores. The NR cohort had a statistically significantly lower ‘relations’ score compared to the two most affected interference categories (p = 0.0113, p = 0.0214), whereas the R cohort had no statistically significant difference between the highest affected categories and ‘relations’, suggesting ‘relations’ were disproportionally unaffected in the NR cohort. The time between visits, sex, age, mutations, pain location and joint replacement presence were not statistically significantly different between the two cohorts. Conclusion Patients with high levels of pain benefited from the service. Factors associated with response were high initial pain scores, use of oral NSAIDs, and a ‘relations’ score which was not statistically significantly different to the other life quality categories. This information could be used in the future to help predict response to pain management interventions in Stickler syndrome. Disclosure R. Mudkavi: None. N. Shenker: None.

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