Chronic non-malignant pain is a worldwide public health problem generally defined as current pain that has lasted for minimum six months or longer, is due to non-life-threatening causes, which has not responded positively to available methods of treatment and may continue throughout the individual's life.1 Self-management is, therefore, a necessity for each individual. In Europe, chronic non-malignant pain affects approximately one out of five people aged 16 years or older.2 It has been well documented that Denmark has a high incidence, with a prevalence of 26.8%, and this trend, based on almost two decades, does not appear to be declining.2-4 Chronic non-malignant pain is often complex, causing difficulty in reaching a diagnosis. The process of diagnosis is naturally prolonged, as several treatment options have to be tested. In addition, it is customary for patients to have to wait for specialized treatment in pain clinics and centers for up to two years, at least in Denmark, due to the lack of general political attention, leading to a lack of overall guidelines and resource priorities in the chronic pain area.5
Chronic non-malignant pain is a chronic illness leading to altered life circumstances, requiring adjustments within the family according to roles and tasks due to the patient's functional impairment and fatigue, and, commonly also, to the occupational situation and social activities of the individual with pain.6,7
Multi-disciplinary chronic pain management programs are led by an interdisciplinary team consisting of at least three of the following professionals: physicians, nurses, physiotherapists, occupational therapists, social workers and psychologists.1,2,8 Due to the complexity of the condition, a patient-centered or biopsychosocial approach is mainly used in an individual and/or group-based context with elements of physical training, cognitive behavior therapy, mindfulness, relaxation training techniques, coping strategy training, workability assessment and others.1,2,8
In recent years, the involvement of relatives in rehabilitation processes has been increasingly investigated as part of a strategy to support the patient's manageability and self-management of chronic disease,9 stroke,10 cancer11,12 and pressure ulcers.13 Research suggests that the involvement of significant others in the rehabilitation process of chronic non-malignant pain produces a positive effect on comprehensive pain management, self-efficacy, level of functioning, stress and common understanding.7,8,14,15 A qualitative chronic pain study based on semi-structured interviews with patients and significant others found that the involvement of significant others in rehabilitation programs may be essential in developing a shared understanding of the management of the condition for those suffering chronic pain, as well as the significant others and the family as a whole.8 Particularly, involvement of significant others seems to be important for the process of realizing and adjusting to the new life circumstances, roles and responsibilities in the home, as well as celebrating achievements together during rehabilitation.8 Another perspective of involving significant others was found in a study exploring the support person's perceptions and experiences of the roles they undertook throughout the rehabilitation process.15 The researchers found that involvement of significant others was entirely dependent on the willingness of the person with chronic pain to involve them. It is also dependent on the extent to which the individual with pain was able to make use of the support provided by their significant other. The main benefits from involving significant others from the significant others point of view were increased understanding of the condition and mechanisms of rehabilitation and therefore feeling more confident in encouraging their loved one to continue training, despite increased levels of pain. In addition, being able to talk with health professionals attuned unrealistic expectations toward rehabilitation.15 In a one-year randomized controlled trial study of 36 chronic low back pain patients and their spouses, it was found that a brief and low-intensity spouse-assisted multi-disciplinary pain management program showed promising results at the 12-month follow-up time point. Patients receiving the spouse-assisted multi-disciplinary pain management program had significant improvements in kinesophobia (fear avoidance behavior) and rumination about pain (the compulsively focused attention on the symptoms of one's distress and on its possible causes and consequences, as opposed to its solution) compared to the two other groups, a patient-oriented multi-disciplinary approach and standard medical care.14
Because of the promising reports on the increased self-management due to involvement of significant others in chronic pain programs,8,15 it is important to conduct a systematic review on the effect of involving significant others in chronic pain programs.
The rationale of this systematic review is to identify the evidence base regarding involving significant others in chronic pain programs and healthcare regimens of adult patients with chronic non-malignant pain in primary, secondary or tertiary healthcare settings. This will inform the development of future case management interventions at multi-disciplinary pain clinics. Developing effective interventions involving significant others is essential, as the presence of chronic pain can affect the family.8,15,17 Accordingly, involvement of significant others is vital to successful comprehensive pain management.7,8,14-17 This review will contribute by drawing attention to the crucial intervention of incorporating significant others in effective and meaningful ways in future rehabilitation programs of chronic non-malignant pain. The following sources were initially searched for published systematic reviews on the topic: JBI Database of Systematic Reviews and Implementation Reports, the Cochrane Library and MEDLINE. No systematic reviews on this topic were found.
Types of participants
The current review will consider studies that address involvement of significant others and include adult patients treated at primary, secondary or tertiary healthcare institutions for chronic (six months or longer) non-malignant/non-cancer pain. Since chronic pain affects adults of all ages, there will be no upper age limit set; we will include participants aged 16 years or over. Patients with cancer pain will be excluded from this review due to the specific nature and management of cancer pain, which often differs from non-cancer-related causes of chronic pain. In addition, adults who are not treated at primary, secondary or tertiary healthcare institutions for chronic benign pain are also considered beyond the scope of this review. In these settings, the involvement of significant others is likely to require different designs compared to community-dwelling interventions and therefore differ greatly from these.7,8,14 The definition of significant others will be in accordance with Abbasi et al.,14 Lewandowski et al. 7 and Swift et al.,8 therefore defined as “people that an individual with chronic pain defines as their key source of social and psychological support”.8 (p.48)
Types of interventions
The current review will consider studies that evaluate any intervention of involving significant others with the purpose of supporting the patient in managing chronic non-malignant pain. Any intervention (mono- or multi-faceted) that includes a defined element of involvement of significant others, delivered to groups or individuals in any format, including face-to-face, telephone and internet-based interventions, will be considered for inclusion. Intervention studies without a defined element of involvement of significant others will be excluded. Significant others may have been selected by the patient, self-selected or selected by health professionals; comprising any relative, spouse, family, friend or neighbor and others. This review will consider studies that have compared involvement from significant others to any intervention that provides usual care as their comparator or other self-management interventions without an element of involving significant others.
The current review will consider studies that include one or several of the following outcome measures of physically or psychological function, self-efficacy or self-assessed pain. The outcomes measures may be, but are not limited to, the Stanford Health Assessment Questionnaire, The Pain Disability Index, Pain Self-Efficacy Questionnaire, The general self-efficacy questionnaire, Arthritis Self-Efficacy Scale, The Pain Stages of Change Questionnaire, the Oswestry Low Back Pain Disability Questionnaire, Occupational Understanding of Challenges – Chronic Pain Questionnaire, The McGill Pain Questionnaire and The Behavioral Assessment of Pain Questionnaire and so on will be considered for inclusion. This is aligned with recommendations for outcome measurement in chronic pain and self-management research.16,18-21 Therefore, these measures are expected to be available for consideration.
Types of studies
The current review will consider both experimental and epidemiological study designs including randomized controlled trials, non-randomized controlled trials, quasi-experimental, before-and-after studies, prospective and retrospective cohort studies, case-control studies and analytical cross-sectional studies for inclusion.
The current review will also consider descriptive epidemiological study designs including case series, individual case reports and descriptive cross-sectional studies for inclusion in a narrative summary. This will enable the identification of current best evidence regarding interventions addressing involvement of significant others for hospitalized adult patients with chronic non-malignant pain.
The search strategy aims to find both published and unpublished studies. A three-step search strategy will be utilized in this review. An initial limited search of MEDLINE and CINAHL will be undertaken followed by analysis of the text words contained in the title and abstract, and of the index terms used to describe the article. A second search using all identified keywords and index terms will then be undertaken across all included databases. Third, the reference list of all identified reports and articles will be searched for additional studies. Studies published in English, German, Danish, Swedish and Norwegian will be considered for inclusion in this review. Studies published from inception of each database until the present will be considered for inclusion in this review.
The databases to be searched include:
Science Citation Index
The search for unpublished studies will include:
Networked Digital Library of Theses and Dissertations
PubMed “related articles” feature will be used to identify any additional studies identified by other sources.
Initial keywords to be used will be:
chronic nonmalignant pain OR chronic non-cancer pain combined with the terms chronic pain program, rehabilitation, self-management, self-efficacy, coping, involvement, participation, spouse-assisted, significant other, family, spouse, effect, quantitative research, quantitative evaluation. Wildcard (*) will be used to ensure all inflections of the word are included in the search. In addition, specific measuring tools will be included in the search: Health assessment Questionnaire, Pain Self-Efficacy Questionnaire, The general self-efficacy questionnaire, Arthritis Self-Efficacy Scale, The Pain Stages of Change Questionnaire, the Oswestry Low Back Pain Disability Questionnaire, Occupational Understanding of Challenges – Chronic Pain Questionnaire, The McGill Pain Questionnaire and The Behavioural Assessment of Pain Questionnaire and Health Education Impact Questionnaire.
Assessment of methodological quality
Papers selected for retrieval will be assessed by two independent reviewers for methodological validity prior to inclusion in the review using standardized critical appraisal instruments from the Joanna Briggs Institute Meta-Analysis of Statistics Assessment and Review Instrument (JBI-MAStARI) (Appendix I). Any disagreements that arise between the reviewers will be resolved through discussion or with a third reviewer.
Data will be extracted by two independent reviewers from papers included in the review using the standardized data extraction tool from JBI-MAStARI (Appendix II). The data extracted will include specific details about the interventions, populations, study methods and outcomes of significance to the review question and specific objectives.
Whenever possible, quantitative papers will be pooled in statistical meta-analysis using JBI-MAStARI. All results will be subject to double data entry. Effect sizes expressed as odds ratio (for categorical data) and weighted mean differences (for continuous data) and their 95% confidence intervals (CIs) will be calculated for analysis. Heterogeneity will be assessed statistically using the standard chi-square and also explored using sub-group analyses based on the different quantitative study designs included in this review. For observational studies, effect sizes expressed as relative risk (cohort studies) and odds ratio (case-control studies) for categorical data and weighted mean differences (continuous data) and their 95% CIs will be calculated for analysis. Where statistical pooling is not possible, the findings will be presented in narrative form including tables and figures to aid in data presentation where appropriate.
Appendix I: Appraisal instruments
MAStARI appraisal instrument
Appendix II: Data extraction instruments
MAStARI data extraction instrument
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