Study Design
We conducted a single centre 8-week pilot intervention study to assess the feasibility and acceptability of the MSC program among adults with OI patients and co-occurring chronic pain. A standardized battery of assessments was completed at baseline and 1-week post MSC intervention, as detailed below. The study was reviewed and approved by the Children’s Hospital of Eastern Ontario (CHEO) Research Ethics Board.
Participants & Recruitment
We recruited participants (≥ 18 years) by screening medical records of former patients seen at CHEO and who had transitioned to adult care between September 2019 and December 2023, as well as their family members with a clinical diagnosis of OI confirmed in their own medical charts. We obtained informed consent either virtually via REDcap (Research Electronic Data Capture) platform (18, 19), verbally over the phone, or in-person.
Participants were recruited if they met the following criteria: Age ≥ 18 years, diagnosed with OI (any type), chronic pain that has been present for at least three months (defined as having pain on most days in the previous three months), able to attend at least 6 of the 8 scheduled MSC program sessions, have a regular health care provider so that proper follow-up can be arranged if participants disclosed high psychological distress during the study (i.e., family doctor, endocrinologist, etc.), and able to provide informed consent. Participants were excluded from the study if they met any of the following criteria: do not speak English with enough fluency to complete all study-related tasks, and active participation in another mental health intervention research trial.
MSC Intervention
The validated MSC intervention (20) consisted of 8 weekly virtual interactive sessions, each lasting for 2 hours (16 hours total). The MSC program encourages and teaches the participants to be open to, rather than disconnected from, their own suffering and treating themselves with the same care they would show to a friend. All participants were asked to keep their cameras on for the duration of the sessions to help create a sense of community, facilitate group interaction, and ensure participant safety. The trial coordinator provided weekly reminders for the duration of the program, via SMS message or email, depending on participant preference, to maximize participant engagement. Each session of the MSC program focused on a specific topic (Table 1). Two trained MSC instructors (one of which was CP) led the program.
Table 1
Session | MSC Intervention Topic | Example Exercises: |
Week 1 | Introduction to MSC: what is MSC, misgivings of self-compassion, physiology of self-compassion and self-criticism | Self-compassion break; soothing and supportive touch |
Week 2 | Practicing Mindfulness: wandering mind, resistance | Affectionate breathing meditation; soles of the feet |
Week 3 | Practicing Kindness: kindness and compassion; loving phrases practice | Affectionate breathing, loving kindness phrases, loving kindness meditation |
Week 4 | Discovering Your Self-Compassionate Voice: stages of progress; self-criticism and safety | Loving kindness for ourselves; motivating ourselves with self-compassion |
Week 5 | Living Deeply: core values, finding hidden value in suffering | Giving and receiving compassion; compassionate listening |
Week 6 | Meeting Difficult Emotions: stages of acceptance; approaches to difficult emotions | Loving kindness for ourselves; being with difficult emotions |
Week 7 | Exploring Challenging Relationships: challenging relationships, pain of disconnection | Compassionate friend; anger and unmet needs; self-compassion break in relationships; compassion with equanimity |
Week 8 | Embracing Your Life: compassion for self and others; maintaining your practice | Compassion for self and others; gratitude for small things; appreciating our qualities |
Data & Outcome Measures
Participants completed a baseline demographic questionnaire (age, marital status, self-reporting gender identity, race/ethnicity, employment status, medical history).
Primary outcomes
Our primary outcomes were feasibility and acceptability of the MSC program. The intervention was defined as feasible if > 75% of participants completed at least 6 of the 8 classes, in line with other studies using the MSC intervention (20) and < 10% of questions from questionnaires left blank. Acceptability of the intervention was assessed by an Intervention Satisfaction Scale, adapted from other studies using the MSC intervention (21), and the Intervention Acceptability Framework (22).
Secondary Outcomes
Our secondary outcomes included a standardized battery of pain, mental health, and general functioning using validated outcome measures as well as assessment of sleep duration and sleep efficiency.
Pain-related outcomes
Pain interference, a measure of the degree of interference that pain has on one’s daily activities, was as assessed by the Patient Reported Outcomes Measurement Information System-Pain Interference (PROMIS-PI) measure (23). The PROMIS-PI provides a score between 0-100, where a score of 50 represents the mean score of a general reference sample, and a score of > 50 represents more pain than that general reference sample. Pain acceptance, a measure of one’s willingness to reduce pain control and focus instead on daily activities and personal goals was assessed by the Chronic Pain Acceptance Questionnaire - Revised (CPAQ) (24). The CPAQ is scored on a scale of 0-120, with higher scores indicating higher levels of acceptance. Pain catastrophizing, which measures magnification, rumination, and helplessness, was assessed by the Pain Catastrophizing Scale (PCS) (25). The PCS is scored from 0–52, with higher scores representing higher pain catastrophizing. Pain severity was assessed by an 11-point scale numerical rating scale ranging from 0 (no pain) to 10 (worst possible pain) (26).
Mental health outcomes
Self-compassion was assessed by the Self-Compassion Scale (SCS) (16). The SCS is scored on a scale of 1–5, with higher scores representing higher self-compassion. Symptoms of depression were assessed by the PROMIS depression tools (27). As with the PROMIS-PI, the PROMIS depression tool measure is scored between 0-100, with 50 representing the mean score for a general reference sample, and scores > 50 representing higher depression than that sample. Emotion regulation, defined as the ability to modulate one’s own emotion and to act in desired ways, was assessed by the Difficulties in Emotion Regulation Scale (DERS) (28). On the DERS, higher scores are suggestive of greater problems with emotion regulation. Experiential avoidance, defined as the tendency to escape or run away from internal experiences (i.e., sensations, emotions, thoughts, memories) that may cause discomfort or suffering, was assessed by the Brief Experiential Avoidance Questionnaire (BEAQ) (29). Higher scores on the BEAQ indicate more experiential avoidance.
General functioning
Days missed from school/work was determined by asking the participants the number of days they missed in the 8 weeks preceding the MSC intervention at baseline, and the number of days missed during the 8 weeks of the intervention. We chose 8 weeks for the timeline of days missed, as to differentiate between baseline and post assessment.
Sleep
Sleep efficiency and sleep duration were objectively measured using GT9X Link watches (ActiGraph Corp, Pensacola, FL, USA) (30). The participants were asked to wear the watch on their non-dominant wrist in the week preceding the intervention, and the last week of the intervention. Participants were asked to track their daily activities, and times they removed the watch on a wear log that was provided to them. They were also asked to complete a daily sleep diary, which tracked the time they went to bed, the time they woke up, and a self-assessment of the subjective quality of their sleep.
Exploratory Outcomes
Our exploratory outcome was the feasibility of collecting objective sleep data using the actigraph watches among adults with OI, defined as the proportion of participants from whom we were able to successfully obtain sleep actigraphy data.
Safety and Risk Considerations
We assessed mental health outcomes through participants self-reports directly into the REDCap database. If a score on the PROMIS Depression scale suggested severe depression (score ≥ 70 (31)), the principal investigator (PI) and study coordinator were alerted. The PI had to contact the participant's primary care provider within 5 days to ensure appropriate follow-up.
Statistical Analysis
Given that this is a pilot study, we chose a convenience sample of 8 adults to allow for an assessment of the program's feasibility.
Descriptive statistics (means and standard deviation [SD] or median, interquartile range [IQR] and range, as appropriate) were used to describe our cohort, the feasibility data, and the patient-reported outcome measures, including intervention satisfaction and acceptability. We explored the impact of attending the MSC program on the various outcome measures using paired t-tests or Wilcoxon tests, as appropriate. Effect size for a given outcome between baseline and 8 weeks was calculated using Cohen’s d for normally distributed data and Cliff’s d for non-parametric data. All tests were two-sided using a 5% significance level.