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Systematic Review and Meta-Analysis

Parent cognitive, behavioural, and affective factors and their relation to child pain and functioning in pediatric chronic pain: a systematic review and meta-analysis

Donnelly, Theresa J.a; Palermo, Tonya M.b,c; Newton-John, Toby R.O.a,*

Author Information
doi: 10.1097/j.pain.0000000000001833

Abstract

1. Introduction

Chronic pain is estimated to affect 25% of children and adolescents.68 A subset of these youth experience impairments in physical and emotional functioning, sleep, quality of life, school attendance, academic performance, peer relations,19,34,60,64,66,77,87,93,95,109,112,139 and an increased risk of pain and psychiatric morbidity in adulthood.41,138,141 The unique developmental influence of parent and family factors on child functioning is integral in understanding the onset, maintenance, and impact of chronic pain in children.73,94,97

Research in pediatric chronic pain has increasingly focused on relationships between parent and family factors and child pain-related experiences, noting consistently observed relationships between specific features of parent and family functioning (eg, parental protective behaviors and family cohesion) with poorer child functioning beyond features of child pain (ie, severity and diagnosis).93,94,114,142 Although the directionality of these relationships remains unclear, the potential to improve child functioning through targeting parent factors is an important avenue for investigation and development of effective interventions.9,35 However, to date, there has not been consistent use of parent interventions,37 and most are focused specifically on modifying parent behaviors rather than addressing other aspects of parent or family functioning. This suggests the importance of a synthesis of the current evidence pertaining to the influence of a broad range of individual parent factors on child pain and related outcomes.

Using a family systems perspective, Palermo and Chambers94 developed a theoretical model that identified specific domains of parent and family variables that are likely to be critical considerations in understanding child pain-related outcomes: individual parent (eg, parenting behaviors and parent affect), parent–child dyadic (eg, parent–child interaction style), and family (eg, family environment) variables. Investigations of individual parent factors largely dominate the literature in this area, with few studies assessing parent–child dyadic or family-level variables. Within the individual parent factors domain, traditional theoretical perspectives such as the operant-behavioral43 and social learning4 models of chronic pain explain connections between parent behaviors and children's pain experiences. For instance, parental reinforcement (eg, through protective or solicitous responses) of child pain behaviors such as withdrawal and avoidance is emphasised in the operant-behavioral model.43 Higher levels of protective behavioral responding are consistently associated with increased disability in children with chronic pain.12,18,97,100,120,142 The social learning perspective highlights the transmission of pain-related cues (eg, cognitive and affective) from parents' own experience of pain50 and their responses to the child in pain.136

While a large number of studies have now investigated associations between various parental factors and child functioning in pediatric chronic pain, no systematic review or meta-analysis exists to summarise available studies or provide estimates of the magnitude of these relationships. The aim of this study was to conduct a systematic review and meta-analysis of modifiable individual parent cognitive, behavioral, and affective factors and to estimate associations with child pain-related variables: pain intensity, disability, school functioning, and psychological functioning.

2. Methods

This systematic review and meta-analysis was conducted in accordance with the PRISMA statement89 PRISMA-P guidelines.116 The review protocol was registered with PROSPERO on 19 January 2018 (CRD 42018081975).

2.1. Search strategy

A search of the electronic databases EMBASE, PsychINFO, Medline, and PubMed was conducted, canvassing records from inception of these databases to February 2019. The central search strategy included key words to identify chronic pain and common chronic pain conditions, pediatric populations, and the inclusion of parent variables (Table 1). Search terms were combined using Boolean logic and searched by keyword to maximize results. A hand search of the reference lists of identified studies was also performed. The search output was imported into Covidence,25 Cochrane's internet-based review platform. After the removal of duplicates, studies were screened by title and abstract by the first author (T.J.D.) to determine which studies clearly did not meet identified inclusion and exclusion criteria. A second reviewer (T.R.O.N.-J.) then also screened these studies with reference to the inclusion and exclusion criteria to determine suitability for inclusion. Differences of opinion were resolved through discussion, and studies meeting all inclusion and exclusion were retained for full-text review. The remaining studies were then subject to full-text review by the first author with the second author providing independent review.

Table 1
Table 1:
Search terms for electronic database search.

2.2. Inclusion criteria

Studies were eligible for inclusion in the current review and meta-analysis if they met the following criteria: (1) published in English in a peer-reviewed journal, (2) included a sample of children and/or adolescents (aged between 3 months and 18 years) with chronic pain (determined either through reference to a recognised chronic or recurrent pain diagnosis, or the reporting of pain exceeding a period of 3 months130), (3) assessed a parental cognitive, behavioral, or affective variable using a valid measure (determined with reference to the availability of some psychometric evidence and measuring a defined construct), and (4) assessed child pain intensity or child pain-related functioning (disability, school functioning, and psychological functioning) using a valid and accepted measure. These criteria were selected to enable the meaningful comparison of results in planned meta-analyses.

2.3. Exclusion criteria

Studies were excluded if they (1) primarily included participants with acute pain or pain duration less than 3 months, (2) included participants with cancer-related or any other life-threatening disease, and/or (3) evaluated an intervention.

2.4. Data extraction

Data were extracted through full-text review of each article by the first author using a standard form designed for the current study. The data extraction form included key study details (author, year, and country), study design, study setting, sample size, age and sex of participants, pain diagnoses, duration of pain, parent sex, parent measure(s), child measure(s), and reported associations between variables of interest including key statistical data (mean, range, correlation coefficient, and P-value). For both parent and child variables, self-report data were prioritized for extraction as self-report measures were most consistently used throughout the literature base. This was to enable appropriate comparisons between like constructs. Where there were parent data available for both mothers and fathers separately, both were extracted.

2.5. Study quality

Data related to the quality of each included study was extracted using the items in the National Institute of Health Quality Assessment Tool for Observational Cohort and Cross-sectional Studies.91 Data related to study population, power, measurement tools, attrition rate, statistical analyses, etc., were collected with reference to each of the 14 items set out in the tool. This was completed independently by 2 authors (T.J.D. and T.R.O.N.-J.), and disagreements were resolved by discussion until consensus was reached.

2.6. Missing data

After full-text review, where studies seemed to have measured variables of interest, however, had not reported key correlational data relevant to the current study, corresponding authors were contacted through email requesting required data. Where no response was received after 2 email attempts, or requested data could not be obtained, the study was excluded.

2.7. Data analysis

To address the study aims of meta-analysing correlations of modifiable parent cognitive, behavioral, and affective factors with child pain variables, the included studies were organised along 2 dimensions: first, through parent domain and, second, with regard to the child variable(s). It was anticipated that the systematic search strategy used would identify a broad number of heterogeneous parental factors within the existing literature. To limit the number of analyses performed, an a priori decision was made to conduct meta-analyses of the correlations with child pain variables for the following individual-level parent variables (those with strong theoretical foundations and evidence base): pain catastrophizing, protective behavioral responses, and psychological functioning (depression, anxiety, and stress associated with parenting a child with pain). These parent variables represent the focus of many existing parent targeted interventions for pediatric chronic pain. Following the grouping of studies into each of these parent domains, studies were then organised with reference to child pain intensity, disability, school functioning, and psychological functioning (anxiety and depression separately). Where there was a minimum of 3 studies reporting correlations between identified variables, a meta-analysis was performed using random-effects models, generating a pooled correlation coefficient. Meta-analyses were conducted using Comprehensive Meta-Analysis Version 311 in which correlations are converted to the Fisher's z scale, and analyses are performed using this index before being converted back to correlations for reporting. Where data were available for both mothers and fathers separately, mothers' data were preferentially included in meta-analyses because this was most comparable with most studies.

3. Results

3.1. Study selection

A total of 12,543 records were identified through electronic database searches. After the removal of 5141 duplicates, screening by title and abstract excluded a further 7069 studies. The full text of the remaining 333 records was reviewed, resulting in 54 studies that met the identified criteria for inclusion in the qualitative synthesis. Of these, 36 studies had data available for inclusion in planned meta-analyses. Figure 1 summarises the PRISMA study selection process.

Figure 1.
Figure 1.:
PRISMA flow chart depicting study selection.

3.2. Missing data

Requests for additional data required to perform the meta-analyses were sent to 8 study authors. Four authors responded providing the requested data, one author responded, however, was unable to provide data as it was no longer available, and 3 authors did not respond to our request.

3.3. Study characteristics

Details of the 54 studies included in this review are summarised in Table 2. Studies originated from 8 countries, the majority being from the United States (n = 45). There were 8819 pediatric participants reported across the studies, ranging in age from less than 1 year to 18 years of age, with a median age of 13.8. Of the studies that reported participant ethnicity (n = 41), participants were overwhelmingly white. Sixty-eight percent of all participants were female, and 87.5% of parents were mothers. Study recruitment occurred primarily in outpatient pain clinics (n = 49), and study samples comprised the following: heterogeneous chronic pain diagnoses (n = 31), rheumatological conditions (n = 13), gastroenterological conditions (n = 4), headache (n = 2), and sickle cell disease (n = 4). Studies were predominantly cross-sectional (n = 50).

Table 2
Table 2:
Characteristics of 54 studies included in systematic review and meta-analysis.
Table 2-A
Table 2-A:
Characteristics of 54 studies included in systematic review and meta-analysis.
Table 2-B
Table 2-B:
Characteristics of 54 studies included in systematic review and meta-analysis.
Table 2-C
Table 2-C:
Characteristics of 54 studies included in systematic review and meta-analysis.
Table 2-D
Table 2-D:
Characteristics of 54 studies included in systematic review and meta-analysis.
Table 2-E
Table 2-E:
Characteristics of 54 studies included in systematic review and meta-analysis.
Table 2-F
Table 2-F:
Characteristics of 54 studies included in systematic review and meta-analysis.

3.4. Measurement of parent variables

Individual parent variables included cognitive, behavioural, and affective variables. Parent cognitions were examined in 20 studies. Pain catastrophizing was the most common cognitive variable assessed (k = 15), measured using the Pain Catastrophizing Scale—Parent Version49 (k = 14) and Bath Adolescent Pain—Parental Impact Questionnaire (BAP-PIQ61; k = 1). Parent fear of pain was assessed using the Fear of Pain Questionnaire—Parent Version121 (k = 2). Other measures used were the Child Vulnerability Scale44 (k = 2), Child Self Efficacy Scale14 (k = 1), Parent Psychological Flexibility Questionnaire84 (k = 2), and Parent Pain Acceptance Questionnaire123 (k = 1).

Parent behavioral responses to child pain were examined in 28 studies. Parent protective or solicitous behaviors were the most prevalent parent behavior assessed (k = 27) using the “protect” subscale of the Adult Responses to Children's Symptoms Scale131 (k = 19), Illness Behavior Encouragement Scale142 (k = 6), Parent Response to Child's Recurrent Pain Episodes Scale143 (k = 1), and Pain-Related Parent Behavior Inventory57 (k = 1). Other parental behavioral responses such as minimizing, punishing, encouraging, etc., were measured in the literature by the ARCS (k = 11), Parent Fear of Pain Questionnaire—“avoidance” subscale122 (k = 2), Parent Responses to School Functioning Scale6 (k = 1), Miscarried Helping for Health Inventory (HIH53; k = 1), and Pain Coping Questionnaire105 (k = 1).

Parent affect or psychological functioning was assessed in 23 studies and reflected significant heterogeneity within the constructs examined. Parent psychological functioning was examined in the most studies (k = 18) and included measures of general parent psychological functioning, parent anxiety, and/or parent depression. Measures used were the Brief Symptom Inventory31 (n = 5), Symptom Checklist-90-Revised30 (n = 3), Health and Daily Living Form90 (n = 2), General Health Questionnaire83 (n = 1), in addition to specific measures of depression and anxiety: Beck Depression Inventory8 (n = 3), Hospital Anxiety and Depression Scale150 (n = 3), and the Spielberger State-Trait Anxiety Inventory125 (n = 1). Other constructs examined included parenting stress specifically associated with caring for a child with chronic pain (k = 8) assessed through the Pediatric Inventory for Parents127 (n = 3), Bath Adolescent PainParent Impact Questionnaire (BAP-PIQ61; n = 1), Pain Experience Questionnaire for ChildrenParent Version57 (n = 1), Child Health Questionnaire—Parent Form 5070 (n = 1), Parent Responses to School Functioning Scale6 (n = 1), and Family Effects of Illness scale126 (n = 1). Studies examining general parenting stress (k = 5) used the Parenting Stress Index78 (n = 3), Stress Index for Parents of Adolescents117 (n = 1), and the Hassles and Uplifts Scale29 (n = 1).

3.5. Measurement of child variables

3.5.1. Child pain intensity

Child pain intensity was reported on in 29 studies, commonly assessed as a single item using a Numerical Rating Scale (n = 14) or Visual Analogue Scale (n = 9). Pain was assessed with reference to current pain (n = 9), average/usual pain (n = 9), highest pain (n = 2), frequency of pain (n = 1), pain unpleasantness (n = 1), or an index reflecting a combination of these. Validated measures assessing the quality of child pain included the Faces Pain Scale—Revised58 (n = 2), Children's Somatization Inventory45 (n = 1), Varni Pediatric Pain Questionnaire135 (n = 1), and Child Health Assessment Questionnaire10 (n = 1).

3.5.2. Child disability and school functioning

Child pain-related disability was assessed in 40 studies, predominantly through the Functional Disability Inventory140 (n = 25). Other measures used were the Child Activity Limitations Interview99 (n = 4), Pediatric Quality of Life Inventory133 (n = 3), Child Health Questionnaire70 (n = 2), BAP Questionnaire (BAPQ36; n = 2), Juvenile Arthritis Functional Adjustment Report—Child Questionnaire59 (n = 2), PROMIS Pain Interference scale134 (n = 1), Pediatric Pain Disability Index135 (n = 1), Modified Fibromyalgia Impact Questionnaire—Child Version113 (n = 1), and Illness Intrusiveness Scale32 (n = 1).

School functioning was assessed through a validated measure in only 3 studies. The PedsQL “school functioning” subscale133 was exclusively used for this purpose.

3.5.3. Child psychological functioning

Child psychological functioning was assessed in 29 studies. Measures used were the Child Depression Inventory69 (n = 13), Revised Manifest Children's Anxiety Scale108 (n = 6), BAPQ “depression” subscale36 (n = 4), BAPQ “general anxiety” subscale36 (n = 3), Child Behaviour Checklist2 (n = 3); Spence Children's Anxiety Scale124 (n = 2), Revised Child Anxiety and Depression Scale15 (n = 1), Behavioural Assessment System for Children107 (n = 1), Health and Daily Living form90 (n = 1), Depression Test for Children (DTC110; n = 1), Children's Anxiety Test-II128 (n = 1), Positive and Negative Affect Scales145 (n = 1), Centre for Epidemiological Studies Depression Scale103 (n = 1), Children's Global Assessment Scale115 (n = 1), and Childhood Psychopathology Measurement Scale82 (n = 1).

3.6. Quality of included studies

Overall, the methodological quality of the included studies was limited by the fact that most studies used a cross-sectional design, thus inherently lowering the quality of evidence. These studies performed poorly on items assessing participant follow-up, blinding of assessors, and sufficient timeframe to see an effect. A number of studies omitted relevant data for the determination of study quality (eg, power analyses) (Fig. 2).

Figure 2.
Figure 2.:
Summary of quality assessment item ratings of studies included in review. CD, couldn't determine; NA, not applicable.

3.7. Parent Factors

3.7.1. Parent cognitions

3.7.1.1. Meta-analyses of parent pain catastrophizing and child pain variables

Correlations between parent pain catastrophizing and child variables were available for 11 studies examining associations with child pain, 12 studies examining associations with child disability, and 3 studies examining child depressive symptoms. There were insufficient studies reporting on the association with child school functioning (k = 1) or child anxiety (k = 1) to perform meta-analyses for these variables.

3.7.1.1.1. Parent pain catastrophizing and child pain

The correlation coefficients of individual studies ranged from r = 0.02 to 0.29 (see Supplementary Fig. 3, available at http://links.lww.com/PAIN/A963). The point estimate of pooled correlations between parent pain catastrophizing and child pain was small (r = 0.16, 95% confidence interval (CI) = 0.12-0.2). This correlation was nevertheless positive indicating that higher levels of parent pain catastrophizing were associated with higher child-reported pain intensity. Results of all meta-analyses can be found in Table 3 (forest plots for all meta-analyses are available at http://links.lww.com/PAIN/A963).

Table 3
Table 3:
Summary of meta-analyses of correlations between parent factors and child pain-related outcomes.
3.7.1.1.2. Parent pain catastrophizing and child disability

Correlation coefficients of individual studies ranged from r = 0.11 to 0.53. The point estimate of pooled correlations between parent pain catastrophizing and child disability was small (r = 0.29, 95% CI = 0.21-0.36). Again, this correlation was positive, indicating that higher parental pain catastrophizing was associated with higher levels of child functional impairment.

3.7.1.1.3. Parent pain catastrophizing and child depression

The correlation coefficients ranged between r = 0.18 and 0.28. The pooled estimate of the correlations was small (r = 0.22, 95% CI = 0.16-0.29), with higher parent pain catastrophizing associated with increased child depressive symptoms.

3.7.2. Parent behaviors

3.7.2.1. Meta-analyses of parent protective behaviors and child variables

Correlations for the subcategory of parent protective behavior were available for 17 studies examining associations with child pain, 22 studies examining associations with child disability, 3 studies examining associations with child school functioning, 9 studies examining associations with child depression, and 7 studies examining associations with child anxiety.

3.7.2.1.1. Parent protective behaviors and child pain

The correlations ranged between r = 0.01 and 0.26 (Lynch-Jordan et al.80 used an updated factor structure for the ARCS and reported correlations between both “protect” and “solicitous” subscales with child pain separately. A decision was made to include the protect subscale only in the meta-analysis). The pooled estimate of the correlations was very small (r = 0.08, 95% CI = 0.01-0.14) demonstrating a positive association between increased protective parent behaviors and increased child pain intensity.

3.7.2.1.2. Parent protective behaviors and child disability

The correlations ranged from r = 0.02 to 0.50 (As outlined above, for Lynch-Jordan et al.80 ibid. the data included in the meta-analysis is the association between the “protect” subscale of the ARCS and child disability). The pooled estimate of the correlations was small (r = 0.25, 95% CI = 0.18-0.31), with higher parent protective behaviors associated with increased child-reported disability.

3.7.2.1.3. Parent protective behaviors and child school functioning

The correlation coefficients ranged from r = −0.24 to −0.54. The pooled estimate of the correlations was medium (r = −0.39, 95% CI = −0.54 to 0.21), with higher parent protective behaviors associated with poorer child-reported school functioning.

3.7.2.1.4. Parent protective behaviors and child depression

The correlations ranged from r = 0.02 to 0.27. The pooled estimate of the correlations was small (r = 0.11, 95% CI = 0.03-0.19) indicating that increased parent protective behaviors was weakly associated with increased child reported depressive symptoms.

3.7.2.1.5. Parent protective behaviors and child anxiety

Correlation coefficients for individual studies ranged from r = 0.04 to 0.25. The pooled correlation coefficient was not significant (r = 0.08, 95% CI = −0.05 to 0.20).

3.7.3. Parent affect

3.7.3.1. Meta-analyses of parent psychological functioning and child pain variables

Correlations for parent depression were available for 4 studies examining associations with child pain, 4 studies examining associations with child disability, 4 studies examining associations with child depression, and 3 studies examining associations with child anxiety. Correlations for parent anxiety were available for 3 studies examining associations with child pain and 4 studies examining associations with child disability. There were insufficient studies examining the relationship between parent anxiety with either child depression or anxiety, and similarly, there were no studies examining the relationship between parent depression or anxiety with child school functioning.

3.7.3.1.1. Parent depression and child pain

The correlations from individual studies ranged from r = 0.05 to 0.23. The pooled correlation coefficient was not significant (r = 0.06, 95% CI = −0.10 to 0.22) indicating no consistent relationship between parent depression and child pain intensity.

3.7.3.1.2. Parent anxiety and child pain

The correlations for individual studies ranged from r = 0.14 to 0.22. The pooled correlation coefficient was small (r = 0.16, 95% CI = 0.07-0.25) indicating a weak positive relationship between parent anxiety and child pain.

3.7.3.1.3. Parent depression and child disability

The correlation coefficients of available studies ranged from r = −0.01 to 0.31. The pooled correlation coefficient was small (r = 0.23, 95% CI = 0.11-0.33) indicating a positive relationship between increased parent depressive symptoms and higher child-reported disability.

3.7.3.1.4. Parent anxiety and child disability

Correlation coefficients reported in individual studies ranged from r = 0.01 to 0.35. The pooled correlation coefficient indicated a small relationship between increased parent anxiety and higher self-reported child disability (r = 0.24, 95% CI = 0.11-0.37).

3.7.3.1.5. Parent depression and child depression

Correlation coefficients from individual studies ranged from r = 0.10 to 0.38. The pooled correlation coefficient reflected a small association between increased parent depression and child-reported depression (r = 0.23, 95% CI = 0.07-0.38).

3.7.3.1.6. Parent depression and child anxiety

Correlation coefficients ranged from r = 0.08 to 0.30. The pooled correlation coefficient was small (r = 0.19, 95% CI = 0.02-0.34) indicating that higher parent depression was mildly associated with increased child anxiety.

3.7.3.2. Meta-analyses of parenting stress and child pain variables

The relationship between parenting stress (with regard to child health) was examined with child pain in 5 studies, with child disability in 5 studies, with child depression in 3 studies and with child anxiety in 3 studies. There were no studies identified examining the relationship between parenting stress and child school functioning.

3.7.3.2.1. Parenting stress and child pain

Correlation coefficients from individual studies ranged from r = −0.04 to 0.43. The pooled correlation coefficient was r = 0.16 (95% CI = −0.18 to 0.45) indicating no reliable relationship between parenting stress and child pain intensity.

3.7.3.2.2. Parenting stress and child disability

The magnitude of correlation coefficients from individual studies ranged from r = −0.12 to −0.57. The pooled correlation coefficient was r = 0.03 (95% CI = −0.40 to 0.44) indicating no consistent relationship between parenting stress and child disability.

3.7.3.2.3. Parenting stress and child depression

Correlation coefficients from individual studies ranged from r = −0.07 to 0.24. The pooled correlation coefficient was r = 0.15 (95% CI = −0.03 to 0.33) demonstrating no consistent relationship between parenting stress and child depression.

3.7.3.2.4. Parenting stress and child anxiety

Correlation coefficients ranged from r = 0.02 to 0.34. The pooled correlation coefficient was small (r = 0.20, 95% CI = 0.00-0.39) indicating that increased parenting stress was associated with increased child anxiety.

4. Discussion

This is the first systematic review and meta-analysis to examine the relationship between a broad range of individual parent factors and children's pain and functioning in samples with chronic pain. Overall, we found large variation in the relationships between individual parent factors and child pain variables. For example, although child pain intensity was only weakly related to parent factors, child pain-related disability was consistently associated with parent cognitive and behavioral factors. Child emotional functioning was not routinely assessed in this literature. Where it was measured, there were no consistent relationships with parent variables. Pain-related school functioning in children was also infrequently assessed, despite its importance in child development. And yet, where it was included in the study designs, the results were unequivocal—the strongest association was the positive relationship between parental protective behavior and child school functioning. Furthermore, the results of this review identified an emerging trend in examining novel individual parent variables such as psychological flexibility and pain acceptance in paediatric chronic pain research. However, the limited number of studies focusing on these constructs and lack of consistency in child outcome variables examined to date limit our current understanding.

4.1. Summary of main findings

4.1.1. Parent cognitions

Pain catastrophizing was the most frequently assessed parental cognition in the included studies. Meta-analyses found higher pain catastrophizing demonstrated a small but significant association with child disability and child depression, and a weak association with child pain intensity. This is consistent with models in the literature that highlights the potential link between parental thoughts and beliefs and child pain experiences.136 Furthermore, this highlights parent pain catastrophizing as an appropriate and central target for parent-focused interventions aimed to improve child outcomes in pediatric chronic pain.

4.1.2. Parent behaviors

Parent behaviors, specifically protective or solicitous behaviors, were the most frequently measured parent factor in the included studies, which is reflective of the operant-behavioral theoretical tradition that guides much of this research.43 The results of meta-analyses demonstrated a moderate relationship between increased protective parent behaviors with increased child disability and decreased school functioning, weak associations with increased child pain intensity and depression, and no consistent association with child anxiety. Although the literature generally finds positive relationships between protective parenting and child anxiety, the absence of this relationship in this study is likely due to certain methodological factors. For example, measures of parent protective behaviors in the pediatric pain literature are limited and narrowly focused on protective or solicitous responses to pain, which may be why the relationship is stronger with aspects of disability including school functioning. Furthermore, the measurement of general rather than pain-specific anxiety in addition to the heterogeneity of child anxiety measures used may also account for the observed pattern of results. The relationship between parental protectiveness and poorer child functioning is largely consistent with theoretical expectations and replicates findings from other pediatric chronic illness populations.12,47,100,148 Again, this finding indicates that targeting protective parent responses to children in pain would likely prove beneficial in parent focused interventions for pediatric chronic pain, which has been studied in a number of pediatric chronic pain interventions.35,98,111

4.1.3. Parent affect

Studies of parent affect examined a diverse range of parental emotional constructs with inconsistent associations observed with child pain variables. Meta-analyses of studies of both parent depression and anxiety demonstrated small but significant associations with child disability; however, surprisingly, studies of parenting stress attributed to parenting a child with chronic pain did not demonstrate a significant relationship. This may be reflective of heterogeneity in the constructs assessed by the measures used in this group of studies. Parent stress attributed to parenting a child with chronic pain was associated only with child anxiety. As expected, parent depression demonstrated a moderate correlation with child depression, replicating findings from other pediatric populations in the literature.7,38,63

Overall, the results of this study can be interpreted within the integrative family systems perspective proposed by Palermo and Chambers.94 These results highlight the influence of individual parent variables identified by operant behavioural and social learning models of pain while also acknowledging the role of broader individual parent variables (ie, affect). The review cannot address the broader family-level variables such as the parent–child dyad and overall family environment because of the very limited studies measuring these domains; thus, we included only individual parent variables.

4.2. Limitations

This review highlights a number of limitations in the current evidence base relating both to methodological weaknesses and restrictions in study scope. Studies reviewed were predominantly cross-sectional and generally used correlational analyses, limiting the quality of data and ability to assess temporal relationships. The heterogeneity of pain conditions comprising study samples should also be considered as a potential limitation. Particularly as research comparing specific parental characteristics in other chronic disease populations (eg, arthritis, sickle cell disease, etc.) have observed differences in patterns of parental distress across disease groups.24

Deficiencies in measures used to assess both parent variables and child pain variables represent another limitation. As has been identified by researchers elsewhere with regard to this literature base,72 assessment of child pain intensity was inconsistent across studies (eg, anchor points, periods, etc.). Measures used to assess parent behaviour were predominantly self-report, which may be influenced by social desirability when completed in clinical settings, potentially obscuring relationships.18

The ability of this review to draw conclusions about the impact of individual parent factors on child school functioning is limited by the paucity of studies that included a validated assessment of school functioning. This has previously been identified as a significant weakness in the literature.48 Although the experience of pediatric chronic pain is widely reported to contribute to both academic and school impairments, no validated measure currently exists to assess all relevant aspects of child school functioning.

Another important limitation is the underrepresentation of fathers in the literature, consistent with historical observations of pediatric health care research.101 Importantly, in studies of pediatric pain where fathers have been included and parental influence analysed by sex, some differences in the pattern of results between maternal and paternal associations with child pain-related outcomes have been observed.75,129,144

An important gap in the literature is understanding of the parent–child dyadic and broader family functioning variables that may also influence child pain. There were also few studies that examined the influence of parental factors by child developmental stage. For example, adolescence is typically a period associated with lessening of parental influence, yet little is known about how this may affect associations between parent factors and chronic pain experiences as children transition to older adolescence. Finally, both the experience of chronic pain and the mechanisms by which parents and families may influence children are known to be influenced by cultural considerations.39 The preponderance of studies originating in North America and comprising predominantly white samples may therefore limit the generalisability of this evidence base to other cultural contexts.

4.3. Future directions

The implications of the review findings and the limitations identified suggest a number of future research directions. First, in addressing methodological limitations evident in the current literature base, longitudinal studies are needed to delineate how relationships between parent and child variables manifest and change over time. Research examining observable variables should seek reports from multiple informants to eliminate potential bias resulting from socially desirable responding, and studies should aim to recruit participants independent of clinical settings to minimise the influence of study setting on participant self-report. In addition, future studies should prioritise the development and utilisation of standardised measures of school functioning.

Second, to identify appropriate targets for intervention, future research should prioritise the inclusion of both mothers and fathers101 and examine relationships separately to delineate parent gender influence, in addition to assessing multiple parent and child variables to elucidate the pathways by which parental factors may exert their influence. Examination of these relationships in the context of child developmental stage, specific pain diagnosis, and cultural context would also likely improve the generalisability of study findings. Furthermore, research examining novel parent factors (eg, psychological flexibility) as well as parent–child dyadic and family functioning variables and their relationship with child pain and related outcomes should be conducted to inform the development of effective interventions for child pain.

4.4. Conclusion

The findings from the current review support the influence of a wide range of individual parent factors in understanding child pain experiences. Specifically, parent pain catastrophizing and protective behaviors are highlighted as important targets for understanding and improving child pain-related disability and school functioning. Methodological weaknesses remain a significant barrier in this literature, and future research addressing these should be prioritised.

Conflict of interest statement

The authors have no conflicts of interest to declare.

Appendix A. Supplemental digital content

Supplemental digital content associated with this article can be found online at http://links.lww.com/PAIN/A963.

Supplemental video content

A video abstract associated with this article is available at http://links.lww.com/PAIN/A962.

Acknowledgements

This research is supported by an Australian Government Research Training Program Scholarship.

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      Keywords:

      Chronic pain; Pain; Children; Parent; Meta-analysis; Systematic review

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