Promoting Resilience in Stress Management (PRISM) and Clinical-focused Narrative (CFN) Pilot in Adults With Primary Mitochondrial Disease (PMD).

Harnessing Resilience in Adults With Primary Mitochondrial Disease: A Pilot Study Investigating the Feasibility of the Promoting Resilience in Stress Management (PRISM) and Clinical-focused Narrative (CFN) Interventions

The goal of this study is to find the best way to help people with primary mitochondrial disease deal with the stress of their condition, and to help these people be better able to "bounce back," or be resilient. In order to do this, the investigators are going to test two interventions (an intervention means that it aims to change something): Promoting Resilience in Stress Management (PRISM) and clinical-focused narrative (CFN) intervention.

Study Overview

• Status: Completed
• Conditions: Mitochondrial Diseases; Mitochondrial Myopathies; MELAS; Mitochondrial Encephalomyopathies; MERRF
• Intervention / Treatment: Behavioral: Promoting Resilience in Stress Management (PRISM); Behavioral: Clinical-focused narrative (CFN)

Detailed Description

The investigators are going to test two interventions in this pilot study. The first is called Promoting Resilience in Stress Management (PRISM). The second is called clinical-focused narrative (CFN) intervention. These interventions involve talking with the researcher about several specific topics.

Participants will join six virtual study visits. They will be assigned to the PRISM group or the CFN group. During the first study visit, participants will learn about the study and will fill out consent forms if they want to participate. In the next four visits, participants will receive one of the study interventions (PRISM or CFN). Some interventions will be supported with a digital app for participants to track progress and review what was talked about in the intervention. The sixth virtual study visit will be a discussion group that happens a couple months after participants finish the intervention.

Participants will also be sent several surveys to complete. One set of surveys is sent just once after the participant enrolls in the study. Another set of surveys is sent after the participant enrolls and again after the participant finishes the interventions.

The study team will review participants' medical records to confirm they have primary mitochondrial disease and to review the genetic diagnosis. If participants do not have copies of their own medical records or if the study team does not already have access to them, the study team will ask participants to sign a release form to obtain a copy of the medical records and/or genetic testing report.

• Study Type: Interventional
• Enrollment (Actual): 16
• Phase: Phase 2; Phase 1

Contacts and Locations

Study Locations

• United States
  • Pennsylvania
    • Philadelphia, Pennsylvania, United States, 19104
      • Children's Hospital of Philadelphia

Participation Criteria

Eligibility Criteria

• Ages Eligible for Study: Adult; Older Adult
• Accepts Healthy Volunteers: No

Description

Inclusion Criteria:

1. Males or females at least 18 years old
2. Capable of giving consent, or there is a legally authorized representative capable of giving consent on the subject behalf and if appropriate, assent.
3. Able to speak and read English
4. Able to participate in Web- and App-based interventions
5. Confirmed molecular diagnosis of primary mitochondrial disease

Exclusion Criteria:

1. Suspected but no molecular diagnosis primary mitochondrial disease

Study Plan

How is the study designed?

Design Details

• Primary Purpose: Treatment
• Allocation: Randomized
• Interventional Model: Parallel Assignment
• Masking: None (Open Label)

Number of Arms

2

Arms and Interventions

Participant Group / Arm	Intervention / Treatment
Active Comparator: Promoting Resilience in Stress Management (PRISM)	Behavioral: Promoting Resilience in Stress Management (PRISM); This program teaches the four pillars of resilience (stress management, goal setting, cognitive restructuring, and benefit-finding) in a one-on-one training program consisting of four 20-30 minute sessions occurring once weekly.
Active Comparator: Clinical-focused narrative (CFN)	Behavioral: Clinical-focused narrative (CFN); This program consists of questions posed to adults with primary mitochondrial disease at regular mitochondrial medicine center visits. Specific topics will be discussed at each session. This consists four 20-30 minute sessions occurring once weekly.

What is the study measuring?

Primary Outcome Measures

Outcome Measure	Measure Description	Time Frame
Determine mean score on validated scale assessing resilience in those who receive PRISM and those who receive CFN.	The Connor-Davidson Resilience Scale (CD-RISC) is a validated measure to assess resilience. Possible scores range from 0-100 (higher scores reflecting greater resilience). Mean scores will be calculated on scales administered to subjects before the intervention and scales administered after intervention completion.	Up to 3 months after the first intervention
Determine change in mean in scores on validated scale assessing resilience between those who receive PRISM and those who receive CFN.	The Connor-Davidson Resilience Scale (CD-RISC) is a validated measure to assess resilience. Possible scores range from 0-100 (higher scores reflecting greater resilience). Mean scores calculated on scales administered before the intervention will be compared to mean scores on scales administered after completion of the intervention.	Up to 3 months after the first intervention
Determine variability in scores on validated scale assessing resilience between those who receive PRISM and those who receive CFN.	The Connor-Davidson Resilience Scale (CD-RISC) is a validated measure to assess resilience. Possible scores range from 0-100 (higher scores reflecting greater resilience). Variability in scores of scales collected before starting and after completion of the interventions will be calculated.	Up to 3 months after the first intervention
Determine mean scores on validated scale assessing coping in those who receive PRISM and those who receive CFN.	The Family Crisis Oriented Personal Scale (F-COPES) is a validated measure to assess coping. Possible scores range from 30 to 150 (higher scores reflect higher levels of coping and problem-solving ability). Mean scores will be calculated on scales administered to subjects before the intervention and scales administered after intervention completion.	Up to 3 months after the first intervention
Determine change in mean scores on validated scale assessing coping between those who receive PRISM and those who receive CFN.	The Family Crisis Oriented Personal Scale (F-COPES) is a validated measure to assess coping. Possible scores range from 30 to 150 (higher scores reflect higher levels of coping and problem-solving ability). Mean scores calculated on scales administered before the intervention will be compared to mean scores on scales administered after completion of the intervention.	Up to 3 months after the first intervention
Determine variability in scores on a validated scale assessing coping between those who receive PRISM and those who receive CFN.	The Family Crisis Oriented Personal Scale (F-COPES) is a validated measure to assess coping. Possible scores range from 30 to 150 (higher scores reflect higher levels of coping and problem-solving ability). Variability in scores of scales collected before starting and after completion of the interventions will be calculated.	Up to 3 months after the first intervention
Determine mean scores on validated scale assessing quality of life in those who receive PRISM and those who receive CFN.	The 36-Item Short Form Survey Instrument (SF-36) is a validated measure to assess quality of life. Possible scores range from 0-100 (higher scores reflect a more favorable health state). Mean scores calculated on scales administered before the intervention will be compared to mean scores on scales administered after completion of the intervention.	Up to 3 months after the first intervention
Determine change in mean in scores on validated scale assessing quality of life between those who receive PRISM and those who receive CFN.	The 36-Item Short Form Survey Instrument (SF-36) is a validated measure to assess quality of life. Possible scores range from 0-100 (higher scores reflect a more favorable health state). Mean scores will be calculated on scales administered to subjects before the intervention and scales administered after intervention completion.	Up to 3 months after the first intervention
Determine variability in scores on a validated scale assessing quality of life between those who receive PRISM and those who receive CFN.	The 36-Item Short Form Survey Instrument (SF-36) is a validated measure to assess quality of life. Possible scores range from 0-100 (higher scores reflect a more favorable health state). Variability in scores of scales collected before starting and after completion of the interventions will be calculated.	Up to 3 months after the first intervention

Secondary Outcome Measures

Outcome Measure	Measure Description	Time Frame
Explore participant experience with PRISM and CFN and assess their acceptability among adults with primary mitochondrial disease.	Semi-structured interviews will be conducted at discussion groups.	Three months after completing the intervention, discussion groups will occur.

Collaborators and Investigators

• Sponsor: Children's Hospital of Philadelphia
• Collaborators: National Institute of Neurological Disorders and Stroke (NINDS); North American Mitochondrial Disease Consortium (NAMDC)
• Investigators: Principal Investigator: Elizabeth M McCormick, MS, Children's Hospital of Philadelphia

Study record dates

Study Major Dates

• Study Start (Actual): September 22, 2023
• Primary Completion (Actual): February 28, 2024
• Study Completion (Actual): February 28, 2024

Study Registration Dates

• First Submitted: September 12, 2023
• First Submitted That Met QC Criteria: September 12, 2023
• First Posted (Actual): September 25, 2023

Study Record Updates

• Last Update Posted (Actual): November 29, 2024
• Last Update Submitted That Met QC Criteria: November 26, 2024
• Last Verified: November 1, 2024

More Information

Terms related to this study

• Additional Relevant MeSH Terms: Musculoskeletal Diseases; Brain Diseases; Central Nervous System Diseases; Nervous System Diseases; Muscular Diseases; Neuromuscular Diseases; Metabolic Diseases; Brain Diseases, Metabolic; Mitochondrial Diseases; Mitochondrial Myopathies; Mitochondrial Encephalomyopathies
• Other Study ID Numbers: 22-020222; U54NS078059-12 (U.S. NIH Grant/Contract)

Plan for Individual participant data (IPD)

• Plan to Share Individual Participant Data (IPD)?: YES
• IPD Plan Description: Data from this study, without direct patient identifiers will be released to an NIH data repository for the purpose of facilitating data sharing according to NIH policy
• IPD Sharing Time Frame: By the time of publication
• IPD Sharing Access Criteria: Anonymized participant data will be released to an NIH repository and will be available upon request.

Drug and device information, study documents

• Studies a U.S. FDA-regulated drug product: No
• Studies a U.S. FDA-regulated device product: No