Self-management Training for Parents With Chronic Muscular Dystrophia (ZRM-CMD-P)

The relentless progressive process of muscular dystrophy requires extraordinary medical, physical, and emotional care with severe consequences for caring parents (increased stress and diminished social, psychological and physical well-being). Despite the obvious need of support for parents only few and weak data exist regarding efficiency and efficacy of specific interventions supporting parental resilience and coping strategies. The presenting study aims to fill this gap by evaluating the efficacy of a structured self-management training for parents of children with severe progressive muscular dystrophy compared to parents receiving treatment as usual (TAU). In addition, investigators measure established biomarkers of psychosocial stress, such as pro-inflammatory cytokines, which will be used to monitor physiological changes with assumed significance for parental health.

Study Overview

• Status: Withdrawn
• Conditions: Muscular Dystrophies
• Intervention / Treatment: Other: Structured self-management-training

Detailed Description

Background:

The relentless progressive process of muscular dystrophy requires extraordinary medical, physical, and emotional care. As a consequences caring for a patient with a severe chronic illness has been associated with increased stress and diminished social, psychological and physical well-being in parents. According to Thompson et al. 57% of parents with Duchenne muscular dystrophy children have self-reported poor psychological adjustment and, even in comparison with parents of children with other burdens like cerebral palsy or renal diseases, muscular dystrophy was associated with a wider spectrum of problems and parental stress. Moreover, parents are shown to have significant stress by feelings of guilt and associated difficulty discussing death issues with their children.

However, despite the obvious need of support for parents only few and weak data exist regarding efficiency and efficacy of specific interventions supporting parental resilience and coping strategies. In practice, they are mostly limited to meetings of self-help groups and casual exchange between concerned parents, which is rather a problem-centered than proactive solution-focused approach. As a consequence, investigators see a great need for studies regarding more specific interventions supporting parents' self-management skills, coping strategies and competencies.

Objectives:

The present study evaluates the efficacy of a structured self-management training for parents of children with severe progressive muscular dystrophy compared to parents receiving treatment as usual (TAU). In addition, investigators measure established biomarkers of psychosocial stress, such as pro-inflammatory cytokines, which will be used to monitor physiological changes with assumed significance for parental health.

Methods:

Participants will fill out online-questionnaires before, during and after self-management training. Training interventions will take place in Zurich as group sessions (10-20 participants per group). Investigators aim for a total of 60-80 participants. Parent-couples or single participants will be randomized in an intervention group (participants receiving self-management training) and a non-intervention group (participants continue with already established support). After one year parents from the non-intervention group will change into the intervention group with self- management training as well. Questionnaires focus on assessing parental strain and self- efficacy and will take 30-45minutes for each of four surveys. Validated biological markers such as cumulative cortisol levels in hair and pro-inflammatory cytokines in the blood will be collected before and after self-management trainings.

• Study Type: Interventional
• Phase: Not Applicable

Participation Criteria

Eligibility Criteria

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

Description

Inclusion Criteria:

• Parents with at least one living child affected by chronic muscular dystrophy

Exclusion Criteria:

• Dying of a child during intervention and analysis.
• Severe/significant psychological disorder (in case of existing psychological support, participation should be agreed/supported by therapist, too)
• Recent or concomitant self-management training

Study Plan

How is the study designed?

Design Details

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

Number of Arms

2

Arms and Interventions

Participant Group / Arm	Intervention / Treatment
Experimental: Intervention; Structured self-management-training (incl. systemic analysis, coaching, teaching) in 4 sessions.	Other: Structured self-management-training; Structured self-management-training (incl. systemic analysis, coaching, teaching) in 4 sessions
No Intervention: Control group; no intervention other than existing support (see exclusion criteria). However, intervention will be offered and studied secondary as well (after 1 1/2 year of no intervention).

What is the study measuring?

Primary Outcome Measures

Outcome Measure	Measure Description	Time Frame
Action Orientation Questionnaire (HAKEMP)	Kuhl and co-workers introduced a special questionnaire (HAKEMP) to assess the tendency of people in concrete situations to revert to an action-oriented or state-oriented condition in 24 items.	1 1/2 year with control group
Self-efficacy: Thought control of action (SWE)	Quantify improved parental resilience and coping strategies facing chronic stress. Questionnaire developed by Jerusalem and Schwarzer, 1981, revised 1999 to test general self-efficacy in 10 items.	1 1/2 year with control group
Change in Experiencing and Behaviour post-intervention (VEV)	VEV is a questionnaire developed by Ziele and Kopf-Mehnert, 1978 to test post-intervention change in experiencing and behavior along with a retrospective life-event checklist and a self-rating questionnaire in 42 items. Other than previous questionnaires VEV will be applied only after intervention.	1 1/2 year with control group

Secondary Outcome Measures

Outcome Measure	Measure Description	Time Frame
Number of Participants With Sum of Abnormal Laboratory Values Concerning Biomarkers of psychosocial stress	Significant lower levels of biomarkers of psychosocial stress, i.e. pro-inflammatory cytokines, which can be used to monitor physiological changes with assumed significance for parental health: C-reactive Protein (hsCRP), Tumor-Necrose-Factor alpha (TNFα), Interleukin (IL)-1ß, IL-6, und Interferon Gamma (IFNγ). Items will be aggregated as a numerical factor which shows the difference of a measured value from a reference value . Abnormal laboratory values therefore will be used as a sum.	1 1/2 year with control group

Collaborators and Investigators

• Sponsor: University Children's Hospital, Zurich
• Collaborators: University of Zurich
• Investigators: Principal Investigator: Jürg C Streuli, MD PhD, University Children's Hospital, Zurich

Study record dates

Study Major Dates

• Study Start: January 1, 2017
• Primary Completion (Anticipated): December 1, 2018
• Study Completion (Anticipated): December 1, 2019

Study Registration Dates

• First Submitted: November 9, 2015
• First Submitted That Met QC Criteria: December 22, 2015
• First Posted (Estimate): December 29, 2015

Study Record Updates

• Last Update Posted (Actual): August 19, 2019
• Last Update Submitted That Met QC Criteria: August 14, 2019
• Last Verified: August 1, 2019

More Information

Terms related to this study

• Keywords: Coping; Muscular Dystrophies; Parental stress; Parental support
• Additional Relevant MeSH Terms: Nervous System Diseases; Genetic Diseases, Inborn; Musculoskeletal Diseases; Muscular Diseases; Neuromuscular Diseases; Muscular Disorders, Atrophic; Muscular Dystrophies
• Other Study ID Numbers: ZRM-CMD Parents