Background
Methods
Protocol and registration
Eligibility criteria, search strategies and screening
Population | Parents/guardians of children (≤18 years of age) with a chronic illness or disability for which the etiology/manifestations are genetic, metabolic or neurologic and specialist pediatric care is required, involving surgical, medical and/or nutritional intervention |
Intervention or exposure | Coping (with or without caregiving complexity) |
Comparator | Quantitative comparison of QoL (outcome) among individuals using different coping strategies or using strategies to different degrees |
Outcome | QoL |
Study characteristics | Peer reviewed, English-language, full text article describing primary study that includes ≥5 participants |
Data extraction and analysis
Study quality assessment
Results
Search and screening
Characteristics of the included studies
Author, year (country) | Study design | Sample size | Illness | Inclusion criteria | Caregiver relation to child | Mean age of caregivers (SD or range) | Age of children |
---|---|---|---|---|---|---|---|
Carona et al., 2014 [24] (Portugal) | Cross-sectional | 156 (epilepsy, n = 65; cerebral palsy, n = 91) | Epilepsy and cerebral palsy | Parent of a child aged 8–18 years who had been diagnosed with epilepsy or cerebral palsy by a physician, and assumed the primary caregiving role at the time of assessment. | Mother (87.7% epilepsy; 90.1% cerebral palsy) | 42.42 years (7.20) epilepsy; 41.47 years (6.26) cerebral palsy | 12.52 years (2.88) epilepsy; 12.07 years (2.82) cerebral palsy |
Dardas & Ahmad, 2015 [23] (Jordan) | Cross-sectional | 184 | Autistic disorder | Parents of children under the age of 12 years with a clinical diagnosis of autistic disorder and could read and write in Arabic. | Mother, 62% | 37 years (SD = 7.6, range from 21 to 69) | 6.3 years (SD = 3, range = 2–12) |
Grey et al., 2011 [63] (USA) | Randomized controlled trial | 123 (coping skills training, n = 75; group education, n = 48) | Type 1 diabetes | Parent of a child who had been diagnosed with Type 1 diabetes for at least 6 months and between the ages of 1–12 years. | 95% mothers, 3% fathers; 2% guardians (female) | 37.3 years (SD = 5.6, range = 26–51) | 8.1 (2.9) coping skills training; 7.9 (2.8) group education |
Guillamon et al., 2013 [48] (Spain) | Cross-sectional | 62 | Cerebral palsy | Father or mother of a child with cerebral palsy (aged less than 18 years) and was the main caregiver. | Mother, 88.7% | 40.95 years (SD = 0.79, range = 29.53) | 7.69 years (SD = 0.19, range = 1–17) |
Hamama-Raz & Hamama, 2015 [60] (Israel) | Cross-sectional | 48 | Epilepsy | Parents of children between 6 and 19 years of age, with Hebrew speaking ability, with only minor learning difficulties, with 1–4 seizures a year, and with the absence of other chronic illnesses. | Mother, 85.42% | 42.90 years (SD = 6.20) | 13.71 years (SD = 3.02, range = 8–18) |
Khanna et al., 2011 [52] (USA) | Cross-sectional | 304 | Autism | Primary caregivers of children with autism aged less than or equal to 18 years of age and had no more than one child diagnosed with autism. | Female, 93.1%; relationship not stated | 38.9 years (SD = 8.0) | 7% < 5 years of age; 44.1% 5-less than 10 years; 41.4% 10-less than 15 years; 6.6% 15–18 years of age |
Khanna et al., 2013 [59] (USA) | Cross-sectional | 316 | Autism | Primary caregivers of children with autism who are aged 18 years or younger and have only one child with autism. | Mother, 91.5% | 18–44 years, 69%; 45–64 years, 30.1% | < 5 years, 16.8%; 5–10 years, 46.5%; 11–18 years, 36.1% |
Motaharian et al., 2015 [53] (Iran) | Cross-sectional | 49 | Hemophilia | Primary caregiver (primary responsibility of providing care to child) of a child (less than 18 years of age) with hemophilia. | Male, 71.4% (relation not stated) | 40 years or younger (44.9%); Older than 40 years (55.1%) | Twelve years or younger (46.9%); Older than 12 years (53.1% |
Raina et al., 2005 [54] (Canada) | Cross-sectional | 468 | Cerebral palsy | Primary caregiver who had a child who participated in the Ontario Motor Growth (OMG) study (explored patterns of gross motor development in children with cerebral palsy), lived with the child, and resided in Ontario. | Mother, 89.7% | 40.3 years (SD = 6.72) | 10.6 years (SD = 2.69) |
Streisand et al., 2010 [57] (USA) | Cross-sectional | 278 | Diabetes (type 1 or type 2) | Parent or guardian who was the most informed about the child’s health, child who was less than 18 years of age and was diagnosed with diabetes by a physician. | Mother (biological, step, foster, or adoptive), 100% | Not reported | 12.1 years (SD = 4.3, range = < 1–17) |
Tseng et al., 2016 [49] (Taiwan) | Cross-sectional | 167 | Cerebral palsy | Primary caregiver of a child with cerebral palsy aged 4 to 12 years, diagnosed by a pediatrician, pediatric neurologist, or physiatrist, and without an additional diagnosis of a neurodegenerative disease or psychiatric illness. | Mother, 82.0%; Father, 13.8% | 40.24 years (SD = 5.43) | Range 4–12 years |
Instruments used to measure key constructs
Measurement of quality of life
Measurement tool | Description of the tool | Reviewed studies using this tool |
---|---|---|
Quality of life instruments
| ||
World Health Organization Quality of Life Assessment Questionnaire (WHOQOL-BREF) [47] | Latent variable composed of four subscales (physical, environmental, psychological, and social relationships) for a total of 26 items; higher score indicates better quality of life. Sub scale scores or a total overall scale can be computed. | |
Medical Outcomes Study Short Form 36 Health Survey (SF-36) [55] | Generic measure of health concepts related to functional status and well-being; comprised of 8 domains and provides summary score for physical and psychological well-being. | [54] |
Medical Outcomes Study Short-Form Health Survey version 2 (SF-12v2) [56] | Comprised of 12 items and 8 health concept subscales, also gives a summary score for physical and mental health status. Physical component sumamry (PCS) is made up of the physical functioning, role physical, bodily pain and general health subscales whereas the mental component (MCS) is comprised of the vitality, social functioning, role emotional, and mental health subscales. | |
EuroQoL five-dimensional (EQ-5D) questionnaire [58] | Consists of five health profile domains: mobility, self-care, usual activities, pain/discomfort and anxiety/depression, each domain is assessed using a single item. A visual analogue scale is also used and ask caregivers to rate their current health status (range from 0 to 100). | [59] |
Quality of Life in Pediatric Epilepsy Scale – Parent Form [61] | Comprised of four sub scales: psychological, physiological, functional, and social, with higher score indicating lower QoL. | [60] |
Parents Diabetes Quality of Life Questionnaire [62] | Assesses parents’ perceptions of the impact of diabetes treatment on their general satisfaction with life; comprised of three subscales: diabetes life satisfaction, disease impact, and disease-related worries. | [63] |
Coping instruments
| ||
Coping Health Inventory for Parents (CHIP) [37] | Comprised of 3 subscales: 1) maintaining family integration, cooperation and optimistic definition of the situation; 2) maintaining social support, self-esteem and psychological stability; 3) understanding the healthcare situation through communication with other parents and consultation with the healthcare team. | |
Brief COPE Inventory [33] | Comprised of 14 sub scales with two items each; broadly captures coping methods. Sub scales can be summarized into adaptive or maladaptive coping strategies. | |
Revised Ways of Coping Checklist [31] | Consists of 66 items separated into eight different cognitive and behavioural strategies (sub scales) used to cope with stressful encounters: confrontive coping, distancing, self-controlling, seeking social support, accepting responsibility, escape avoidance, planful problem solving, and positive reappraisal. | [23] |
Calsbeek Coping Inventory for Stressful Situations (CISS) [65] | Comprised of 21 items in three domains: problem-oriented, emotion-oriented, and avoidance-oriented coping strategies. | [53] |
Perceived Ability to Cope with Trauma Scale (PACT) [66] | Comprised of two sub scales: emotional processing and forward focus, single score used. | [60] |
Issues in Coping with IDDM-Parent Scale [67] | Measures parents’ issues in coping with their child’s diabetes; Consists of two sub scales: how difficult and how upsetting parents find it to cope with issues related to management of child’s Type 1 Diabetes. | [63] |
Family Coping Patterns Questionnaire (FCPQ) [68] | Comprised of 34 items in 3 sub scales: avoidance-oriented coping, cognitive appraisal-oriented coping, and task-oriented coping strategies | [49] |
Caregiving complexity instruments
| ||
Revised Burden Measure [74] | Latent variable composed of three subscales: Relationship Burden, Objective Burden, Subjective Burden. | [24] |
Revised Scale for Caregiving Self-Efficacy [75] | Consists of three domains: obtaining respite, responding to disruptive patient behaviours, and controlling upsetting thoughts. Sub scale and overall scores. | [48] |
Caregiver Strain Questionnaire [76] | Consists of three domains: objective strain, subjective internalized strain, and subjective externalized strain, an overall burden score is used. | |
Pediatric Evaluation of Disability Inventory (PEDI) [77] | Measures amount of caregiver assistance provided to a child during basic functional activities of daily living. |
Measurement of coping
Quality assessment
Quality Assessment Criteria | Study | ||||||||||
---|---|---|---|---|---|---|---|---|---|---|---|
Carona et al. (2014) [24] | Dardas &Ahmad (2015) [23] | Grey et al. (2011) [63] | Guillamon et al. (2013) [48] | Hamama-Raz & Hamama (2015) [60] | Khanna et al. (2011) [52] | Khanna et al. (2013) [59] | Motaharian et al. (2015) [53] | Raina et al. (2005) [54] | Streisand et al. (2010) [57] | Tseng et al. (2016) [49] | |
Was the research question or objective in this paper clearly stated? | ✓ | ✓ | ✓ | ✓ | ✓ | ✓ | ✓ | ✓ | ✓ | ✓ | ✓ |
Was the study population clearly specified and defined? | ✓ | ✓ | ✓ | ✓ | ✓ | ✓ | ✓ | ✓ | ✓ | ✓ | ✓ |
Was the participation rate of eligible persons at least 50%? | ✗ | NR | ✓ | NR | ✗ | ✗ | NR | ✓ | ✓ | ✓ | NR |
Were all the subjects selected or recruited from the same or similar populations (including the same time period)? Were inclusion and exclusion criteria for being in the study prespecified and applied uniformly to all participants? | ✗ | ✓ | ✓ | ✓ | ✓ | ✓ | ✓ | ✓ | ✓ | ✓ | ✓ |
Was a sample size justification, power description, or variance and effect estimates provided? | ✗ | ✓ | ✗ | ✗ | ✓ | ✗ | ✗ | ✗ | ✗ | ✗ | ✗ |
For the analyses in this paper, were the exposure(s) of interest measured prior to the outcome(s) being measured? | ✗ | ✗ | ✓ | ✗ | ✗ | ✗ | ✗ | ✗ | ✗ | ✗ | ✗ |
Was the timeframe sufficient so that one could reasonably expect to see an association between exposure and outcome if it existed? | ✗ | ✗ | ✓ | ✗ | ✗ | ✗ | ✗ | ✗ | ✗ | ✗ | ✗ |
For exposures that can vary in amount or level, did the study examine different levels of the exposure as related to the outcome (e.g., categories of exposure, or exposure measured as continuous variable)? | N/A | N/A | N/A | N/A | N/A | N/A | N/A | N/A | N/A | N/A | N/A |
Were the exposure measures (independent variables) clearly defined, valid, reliable, and implemented consistently across all study participants? | ✓ | ✓ | ✓ | ✓ | ✓ | ✓ | ✓ | ✓ | ✓ | ✗ | ✓ |
Was the exposure(s) assessed more than once over time? | N/A | N/A | ✓ | N/A | N/A | N/A | N/A | N/A | N/A | N/A | N/A |
Were the outcome measures (dependent variables) clearly defined, valid, reliable, and implemented consistently across all study participants? | ✓ | ✓ | ✓ | ✓ | ✓ | ✓ | ✓ | ✓ | ✓ | ✗ | ✓ |
Were the outcome assessors blinded to the exposure status of participants? | N/A | N/A | NR | N/A | N/A | N/A | N/A | N/A | N/A | N/A | N/A |
Was loss to follow-up after baseline 20% or less? | N/A | N/A | ✓ | N/A | N/A | N/A | N/A | N/A | N/A | N/A | N/A |
Were key potential confounding variables measured and adjusted statistically for their impact on the relationship between exposure(s) and outcome(s)? | ✗ | ✗ | ✓ | ✓ | ✓ | ✓ | ✓ | ✗ | ✓ | ✓ | ✓ |
Overall Quality | Good | Fair | Good | Good | Good | Good | Good | Poor | Good | Poor | Good |
Findings regarding the association between caregiving complexity and quality of life
Author, year | Variables of interest | General approach to analysis | Main findings |
---|---|---|---|
Carona et al., 2014 [24] | Coping: behavourial disengagement (avoidant, emotion-focused strategy) QoL: overall QoL Caregiving complexity: caregiving burden measured | Structural equation modelling | (1) Caregiving burden directly and negatively predicted parents’ QoL (2) Behavioural disengagement directly and negatively predicted parents’ QoL (3) Caregiving burden had a significant indirect effect on parent’s QoL via behavioural disengagement coping. (4) Coping had a mediating role in the association between caregiving burden and quality of life. |
Dardas & Ahmad, 2015 [23] | Coping: eight sub scale measurements QoL: overall QoL | Bivariate and multivariable regression | (1) Escape avoidance and accepting responsibility coping strategies were significantly and inversely associated with QoL. (2) Accepting responsibility was found to mediate the association between stress and QoL. (3) Escape avoidance and seeking social support were found to moderate the relationship between stress and QoL. |
Grey et al., 2011 [63] | Coping: issues in coping measurement (higher score = coping is more upsetting and difficult) QoL: Disease impact on general life satisfaction (higher score = greater negative impact) | Correlation | (1) While controlling for baseline coping, change in coping at 3 months was not significantly correlated with change in quality of life at 3, 6, or 12 months. |
Guillamon et al., 2013 [48] | Coping: sub scale measurements of integration, social support, and understanding QoL: sub scale measurements of physical, social relationships, environment (considered QoL), and mental health, BDI, and STAI-Trait (considered to be mental health). Caregiving complexity: self-efficacy measured | Multivariable regression | (1) Coping strategies did not significantly predict any quality of life or mental health indicators. (2) Caregiving self-efficacy was a significant, positive predictor of the quality of life environment and mental health sub scales, and significantly predicted caregiver anxiety. |
Hamama-Raz & Hamama, 2015 [60] | Coping: flexibility measurement QoL: sub scale measurements (physical, psychological, social, and functional) as well as overall QoL (higher score = lower QoL) | Correlation and multivariable regression | (1) Flexibility coping was significantly and inversely correlated with each QoL sub scale as well as overall QoL. (2) Flexibility coping was significantly and negatively associated with psychological, functional, and overall QoL (i.e., greater use of flexibility coping was associated with a decreased negative effect of QoL). |
Khanna et al., 2011 [52] | Coping: adaptive and maladaptive sub scale measurements QoL: physical and psychological sub scale measurements Caregiving complexity: caregiving burden measured Disease severity: care recipient functional status | Multivariable regression and structural equation modelling | (1) Care recipient functional status, maladaptive coping, and caregiver burden were significantly and negatively associated with psychological QoL. (2) Maladaptive coping had a direct negative effect on psychological QoL. (3) Adaptive and maladaptive coping had a direct positive effect on caregiver burden which in turn had a direct negative effect on psychological QoL. |
Khanna et al., 2013 [59] | Coping: adaptive and maladaptive sub scale measurements QoL: overall health-related QoL Caregiving complexity: three sub scale measurements- objective strain, subjective internalized strain, and subjective externalized strain Disease severity: parent-reported measurement of social interaction, communication, and restricted and repetitive behavior (overall score given) | Correlation and multivariable regression | (1) Objective and subjective internalized strain were significantly and inversely correlated with QoL. (2) Maladaptive coping was significantly and inversely correlated with QoL. (3) Objective strain was a significantly and negatively associated with QoL. |
Motaharian et al., 2015 [53] | Coping: three sub scale measurements given (problem-, emotion-, and avoidance-oriented) QoL: overall QoL | Correlation and multivariable regression | (1) Emotion- and avoidance-oriented coping was significantly and inversely correlated with QoL. (2) Emotion- and avoidance-oriented coping were significantly and negatively associated with QoL in the regression model. |
Raina et al., 2005 [54] | Coping: stress management QoL: sub scale measurements of physical and psychological QoL Caregiving complexity: caregiving demand | Structural equation modelling | (1) Stress management (coping) had a direct positive effect on caregiver psychological health. (2) Caregiving demand was directly and positively associated with physical and psychological health QoL of caregivers (greater score = less demand). |
Streisand et al., 2010 [57] | Coping: single item, respondents asked how well they felt they were coping with the day-to-day demands of parenthood and raising children. QoL: Parent physical and psychological well-being measured using single item. | Bivariate associations and multivariable regression | (1) Coping was significantly associated with psychological and physical QoL in bivariate and multivariate models. |
Tseng et al., 2016 [49] | Coping: three sub scale measurements given QoL: sub scale measurements of physical, psychological, social relationships, and environment Caregiving complexity: factor analysis of items from two sub scales of the Chinese version of the Pediatric Evaluation of Disability and Inventory (measuring Functional Skills and Caregiver Assistance) Disease severity: scales that measured gross motor impairment severity and fine motor impairment severity | Multivariable regression | (1) Greater use of avoidance-oriented coping positively associated with all QoL domains; perceived helpfulness or use of cognitive appraisal-oriented coping associated with higher psychological and social QoL, respectively; perceived helpfulness of task-oriented coping associated with higher environment QoL (2) Degree of child fine motor impairment associated with physical, social, and environment domains of QoL but not with psychological QoL (3) Caregiving complexity (functional skills and need for caregiver assistance) not included in final (stepwise) multivariable models |