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Catastrophizing, pain, and functional outcomes for children with chronic pain

a meta-analytic review

Miller, Megan M.a; Meints, Samantha M.b; Hirsh, Adam T.a,*

doi: 10.1097/j.pain.0000000000001342
Systematic Reviews and Meta-Analyses
Global Year 2018
Editor's Choice

Pediatric chronic pain is associated with numerous negative outcomes including increased physical disability, increased rates of depression and anxiety, and decreased quality of life (QOL). Pain catastrophizing–broadly conceptualized as including rumination, magnification, and helplessness cognitions surrounding one's pain–has been linked with poor functional outcomes in children with chronic pain. Pain catastrophizing in pediatric chronic pain is often considered a key factor on which to focus treatment efforts. However, absent a systematic review that integrates the relevant literature, this call for routine assessment and targeted treatment may be premature. This study aimed to: (1) meta-analytically quantify the relationship between catastrophizing and pain and functional/psychosocial outcomes (functional disability/physical functioning, anxiety, depression, and QOL) in children with chronic pain, and (2) examine potential moderators of these relationships. Using a random-effects model, a total of 111 effect sizes from 38 studies were analyzed. Effect sizes ranged from medium to large, with anxiety, depression, and QOL demonstrating a strong association with catastrophizing. Pain intensity and physical disability had a moderate association with catastrophizing. These relationships were robust, minimizing potential publication bias. None of the examined moderators were significant. The strong relationships found between catastrophizing and anxiety, depression, and QOL suggest that successfully intervening on catastrophizing could have far reaching implications in improving pain outcomes in pediatric chronic pain.

aDepartment of Psychology, Indiana University-Purdue University Indianapolis, Indianapolis, IN, United States

bDepartment of Anesthesiology, Pain Management Center, Brigham and Women's Hospital, Harvard Medical School, Chestnut Hill, MA, United States

Corresponding author. Address: Department of Psychology, Indiana University-Purdue University Indianapolis, LD124, 402 N Blackford St, Indianapolis, IN 46202, United States. Tel.: (317)-274-6942; fax: (317)-274-6756. E-mail address: athirsh@iupui.edu (A.T. Hirsh).

Sponsorships or competing interests that may be relevant to content are disclosed at the end of this article.

Received March 04, 2018

Received in revised form June 04, 2018

Accepted June 20, 2018

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1. Introduction

Chronic pain is associated with negative outcomes in children and adolescents, including worse physical and emotional functioning.5,9,33,50,53,54,66,75,81,83,100 Many factors have been explored to determine why chronic pain leads to such poor outcomes. Pain catastrophizing is one such factor.19,104 As detailed by Turner and Aaron,113 catastrophizing is historically rooted in the broader psychological literature and is most commonly linked with anxiety disorders12,102 and depression.32 Nearly 2 decades after what many consider to be the first use of the term by Ellis,24 scholars began characterizing and examining the impact of catastrophizing about pain specifically.1,13,14,89,101 Several of the early self-reported measures of pain-related coping, such as the Cognitive Error Questionnaire (CEQ)63 and the Coping Strategies Questionnaire (CSQ),89 included items assessing catastrophic thinking about pain. However, the content of these items differ across measures. Many of the items from the CEQ assess pessimistic pain-related thoughts about the future (eg, “If I do not get some time to relax during the day, I am going to be bedridden and unable to work.”), whereas items from the CSQ focus solely on helplessness cognitions (eg, “I feel like I cannot go on.”). Over 10 years later, in an effort to integrate the various theories and measurements of pain catastrophizing, Sullivan et al.104 created the Pain Catastrophizing Scale (PCS), which includes 3 separate but related domains: rumination, magnification, and helplessness. Although the work of Sullivan et al. mark a turning point in our understanding of pain catastrophizing (at least as indicated by the exponential growth in the literature and the predominance of the PCS as the gold standard measure), Turner and Aaron argue that none of the currently available measures, including the PCS, fully capture the construct of catastrophizing as defined in the broader field of psychology.113 Specifically, they contend that none tap into the thoughts about the worse possible outcomes of pain (eg, paralysis and complete disability), although they do acknowledge that the CEQ comes closest. Given these historical developments, and the fact that the three-factor model is privileged in most of the contemporary work in this area, we adopted the conceptualization of pain catastrophizing by Sullivan et al.104 for the current systematic review.

The aforementioned developments focused exclusively on adults with pain. Nearly a decade later, the pediatric literature began examining the impact of catastrophizing in pediatric pain experience. Self-reported measures assessing pain catastrophizing in children and adolescents were created de novo (eg, Pain Response Inventory [PRI]122 and Pain Coping Questionnaire [PCQ]88) or adapted from existing adult versions (eg, CSQ–Child version35 and PCS–Child version19). Research using these measures has found that catastrophizing in children with chronic pain is linked to greater pain and disability (cross-sectionally and longitudinally),8,19 and to increased pain behaviors74 and analgesic use.7 In addition, previous research found that high catastrophizing children had significantly worse psychological outcomes than low catastrophizing children,21 with high catastrophizers scoring 4 times higher on a measure of depressive symptoms than their counterparts.74

On account of these advancements in the theory and measurement of pain catastrophizing, along with the seemingly consistent pattern of findings suggesting that catastrophizing contributes to negative pain outcomes for children, several scholars have called for routine assessment of pain catastrophizing in pediatric chronic pain and consider it a key factor on which to focus treatment efforts.2,55,128 However, this call for routine assessment and targeted treatment may be premature. To date, no study has systematically integrated the previous results, quantified the magnitude of the relationship between catastrophizing and poor pain outcomes in children, or explored possible moderating factors. Such information is critical for health care providers and administrators to best allocate finite clinical and financial resources to the patients who are most in need.

Given the rapid developmental changes–physical and psychosocial–that take place during childhood and adolescence, several moderators should be examined in the context of catastrophizing and its relation to poor outcomes in pediatric chronic pain. Age is one such moderator. Research suggests that increased age is associated with increased levels of catastrophizing;7 thus, the strength of the relationships between catastrophizing and pain outcomes may vary with age. Sex may also moderate the relationship between catastrophizing and pain outcomes. Socialization in relation to pain differs for girls and boys. In both healthy and chronic pain samples, pain catastrophizing is associated with higher levels of pain expression.117,118 This is potentially important in the context of sex differences, given that girls display more pain behaviors than boys, which may result in girls receiving more comfort from others when in pain.28 This differential reinforcement may, in turn, contribute to sex differences in catastrophizing and pain outcomes.

In addition to developmental factors that may affect the relationship between catastrophizing and pain outcomes, there are several other factors to consider. Pain severity may influence the relationship between catastrophizing and pain outcomes, given that increasing levels of pain are related to higher levels of catastrophizing.94,108 Pain duration may also influence this relationship. A longer pain duration may exacerbate catastrophic and helpless perceptions (ie, “It is never going to get any better”), or it may attenuate them (ie, “I need to accept this pain”). Another clinical factor to consider is type of chronic pain. Research suggests that levels of catastrophizing differ by pain diagnosis. For example, several studies have found that sickle cell patients have higher levels of pain catastrophizing than those with rheumatoid arthritis or musculoskeletal pain.16,47 In addition to developmental and clinical factors, the specific measure used to assess catastrophizing may affect its relationship to pain. Measures of catastrophizing vary across clinical and research settings. These measures have different conceptualizations (unidimensional vs multidimensional) of catastrophizing and consequently, different psychometric properties. These differences between measures may influence the association between catastrophizing and pain outcomes.

In summary, catastrophizing is commonly touted as a key clinical construct to measure and focus treatment efforts on in the pediatric pain context. However, no systematic review has been conducted to integrate the relevant literature and guide these clinical efforts. Filling this knowledge gap would enhance treatment and guide allocation of resources to the patients who would benefit the most from treatment. Thus, the aims of this study were to: (1) meta-analytically quantify the relationship between catastrophizing and pain and functional/psychosocial outcomes (physical functioning, anxiety, depression, and quality of life [QOL]) in children with chronic pain, and (2) examine age, sex, pain intensity, pain duration, pain diagnosis, and measure type as moderators of these relationships.

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2. Methods

2.1. Literature search

Empirical studies were identified using PsycInfo, Medline, PubMed, and Embase databases. Relevant articles published through March 23, 2017, were selected based on searches defined by all possible keyword combinations of terms for (1) catastroph*, (2) child*, youth, adoles*, and (3) pain, chronic pain, nociception. Electronic mail alerts were created using these terms to identify articles published after the initial search. Reference sections of identified empirical studies were reviewed for additional relevant studies. In addition, forward searches were conducted using identified articles to find additional relevant articles. Study authors were contacted for any necessary information needed for analyses from studies that reported insufficient information (Table 1).

Table 1

Table 1

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2.2. Inclusion and exclusion criteria

Studies were included if they (1) had measures of child-reported catastrophizing and one or more of the following: pain intensity, physical functioning, depression, anxiety, or QOL, (2) were based on an independent child or adolescent sample (between the ages of 8 and 21) with chronic pain (ie, persistent pain for 3 or more months), and (3) were available in English. Also, the studies needed to provide an effect size or contain information that allowed for the calculation of an effect size representing the relationship between catastrophizing and one or more of the pain outcomes of interest. Only effect sizes representing the relationship between baseline, or preintervention, levels of catastrophizing and functional/psychosocial outcomes were included.

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2.3. Coding of studies

Each article was read and coded independently by 2 study authors (M.M.M. and S.M.M.) using a standardized coding form.

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2.3.1. Basic study information and study variables

Sample-level information included publication year and type of publication. Sample characteristics included total sample size, race (percent White), sex (percent female), mean age, and whether or not the sample included participants older than 18 years.

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2.3.2. Moderator coding

Sex (percent female), mean age of sample, mean pain intensity rating, and mean pain duration (in months) were coded as continuous moderators. Type of chronic pain and catastrophizing measure were coded as categorical moderators.

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2.4. Quality assessment

Studies were assessed for common sources of bias in observational studies.41,91 Many items on the typical quality rubrics used for meta-analyses are not relevant for correlational designs.44,79 Thus, for the current study, we used a modified version of the rubric from the meta-analysis by Salyers et al.,90 such that study quality ratings (QRs) ranged from 0 to 10. The initial rating system was tested and refined on several studies before applying it to the full sample of studies. Interrater reliability of the initial codes was strong (r = 0.81, P < 0.01), and disagreements were resolved through discussion.

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2.5. Meta-analytic method

2.5.1. Mean effect size

Pearson r was used as the effect size statistic for the relationship between catastrophizing and the pain outcomes of interest. Effect sizes were coded such that higher values reflect higher levels of catastrophizing and greater pain intensity, poorer physical functioning, higher levels of anxiety symptoms, higher levels of depressive symptoms, and worse QOL. All effect sizes were corrected using Fisher r-to-Z transformation, which mitigates the problematic SE formulation inherent in using r in its raw form. Effect sizes at the study level were weighted by sample size to account for the SE in effect size estimates.11 When a study provided multiple values for an association, an average effect size was calculated to reduce bias.11

A random-effects model was used, when appropriate (k ≥ 6), due to effects of both within-study and between-study variability;65 when k <6, the fixed-effects model was reported. Effect sizes, mean effect sizes, and moderation models were calculated using IBM SPSS Statistics 24 and macros provided by Wilson.127 Effect sizes were transformed back to r for ease of interpretation using the inverse of the Fisher r-to-z transformation. Correlation coefficients of less than 0.10 were considered small, correlations of 0.25 were considered medium, and correlations greater than or equal to 0.40 were considered large.65

Heterogeneity for each overall effect size was examined using the Q-statistic,11 with significant results (P < 0.10) suggesting moderation.45 The I2 index was calculated to examine the extent of heterogeneity.45 When I2 values were greater than or equal to 25%, moderator analyses were conducted,49 as this suggests that between-study variability in effect sizes exceeds levels that would be expected by chance.49

A fail-safe N analysis was conducted to estimate how many studies with null findings would be necessary to reduce the effect sizes to nonsignificance.80 Q-test effect size comparisons (with follow-up z tests) were conducted to determine the largest effect size for each outcome. The influence of publication bias was evaluated using Egger regression approach and funnel plots.23

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2.5.2. Moderation analyses

Categorical moderators were tested using Q statistics and I2 indices (polarized effect sizes, decreased I2, and small confidence interval [CI] ranges indicating moderation), and continuous moderators were assessed for significant beta weights and decreased I2 using meta-regressions.49 Because meta-regressions use list-wise deletion, each moderator was examined independently to maximize the number of studies included in the analysis. A minimum of 2 studies per comparison group for categorical moderators was considered necessary for conducting planned meta-analyses.

For the first aim, the mean effect sizes between catastrophizing and pain, physical functioning, anxiety, depression, and QOL were calculated. For the second aim, categorical and continuous moderators were explored when heterogeneity indexes indicated potential moderation.

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2.5.3. Subgroup analyses

In an effort to be inclusive, studies that included participants older than 18 years or a small subset of participants reporting pain for <3 months were included. Differences in mean effect size based on inclusion or exclusion of these samples were explored in subgroup analyses.

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3. Results

3.1. Study sample

One thousand two hundred eighty-two records were identified through the initial database search. Sixty-five studies met inclusion criteria. Of these 65 studies, 36 did not report sufficient effect size information. All corresponding authors were contacted through email to obtain required information. Twelve authors provided the necessary information and these data were included in the final sample, resulting in 38 unique samples of children with chronic pain (Fig. 1 for PRISMA flow diagram). Two studies were included that overlapped with other study samples but reported a unique catastrophizing—pain outcome relationship—and so were retained for analysis. Descriptions of these 2 studies were omitted in the study summary (Table 1) and study quality (Table 2) tables to avoid duplication.

Figure 1

Figure 1

Table 2-a

Table 2-a

Table 2-b

Table 2-b

Table 2-c

Table 2-c

A total of 111 effect sizes from 38 studies were included in the final sample. Each study provided an average of 2.5 effect sizes. Studies used the following self-reported measures of pain catastrophizing: Pain Catastrophizing Scale–Child and adult report (PCS), PCQ, Coping Strategies Questionnaire–Child, PRI, and Pain-Related Cognitions Questionnaire. The most commonly used measures were Pain Catastrophizing Scale–Child (k = 23), PCQ (k = 8), and PCS (k = 5).

The overall sample size contained 6202 participants, with the mean sample size for included studies equal to 77 participants. The study samples were, on average, predominately female (M = 72%) and White (M = 80%). The mean age for the samples was 14.4 years (range: 11.2-18.3). Three studies provided multiple effect sizes for one of the catastrophizing–pain outcome relationships; these effect sizes were averaged within each study for subsequent analyses. Additional study summary characteristics are reported in Table 1.

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3.2. Relationship between catastrophizing and pain outcomes

Table 3 presents the original and corrected (Fisher r-to-Z transformed) effect sizes and sample-level information for studies included in the final meta-analysis.

Table 3-a

Table 3-a

Table 3-b

Table 3-b

Table 3-c

Table 3-c

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3.2.1. Pain intensity

The meta-analysis of the relationship between catastrophizing and pain intensity included 35 independent samples and resulted in a positive medium effect size, with r = 0.29 (95% CI: 0.24-0.34, Table 4). The overall effect was significantly different from zero (z = 11.37, P < 0.01). Orwin fail-safe N analysis indicated that an additional 172 studies with null effects would be needed to reduce the overall mean effect to nonsignificance. Egger regression test of asymmetry resulted in a precision value = −0.17 (90% CI: −1.37 to 1.04, P = 0.78), indicating no significant amount of asymmetry and suggesting no significant amount of publication bias.23 The funnel plot indicates a roughly symmetric distribution of effect sizes, with most of the effects around the mean intercept, further suggesting that publication bias is unlikely (Fig. 2). The Q-statistic of the overall effect was 85.12, with a substantial amount of heterogeneity (I2 = 60.06%), warranting additional moderation analyses.

Table 4

Table 4

Figure 2

Figure 2

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3.2.2. Physical functioning

The meta-analysis of the relationship between catastrophizing and physical functioning included 30 independent samples and resulted in a positive medium effect size, with r = 0.39 (95% CI: 0.35-0.43, Table 4). The overall effect was significantly different from zero (z = 15.26, P < 0.01), and an additional 210 studies with null effects would be needed to reduce this to nonsignificance. Egger regression test of asymmetry resulted in a precision value = −0.17 (90% CI: −1.44 to 1.09, P = 0.78), indicating no significant amount of asymmetry and minimal publication bias (Egger et al., 1997). The funnel plot was roughly symmetrical, further suggesting that publication bias is unlikely (Fig. 3). The results of heterogeneity analyses (Q = 72.33, I2 = 59.91%) supported additional moderation analyses.

Figure 3

Figure 3

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3.2.3. Anxiety

The meta-analysis of the relationship between catastrophizing and anxiety included 15 independent samples and resulted in a positive large effect size, with r = 0.55 (95% CI: 0.50-0.59, Table 4). The overall effect was significantly different from zero (z = 18.96, P < 0.01). Orwin fail-safe N analysis indicated that an additional 165 studies with null effects would be needed to reduce the overall mean effect to nonsignificance. Egger regression test of asymmetry resulted in a precision value = 0.37 (90% CI: −1.00 to 1.73, P = 0.57), indicating no significant amount of asymmetry and minimal publication bias.23 The funnel plot was roughly symmetrical, further suggesting that publication bias is unlikely (Fig. 4). The Q-statistic of the overall effect was 28.04, with a moderate amount of heterogeneity (I2 = 45.91%).

Figure 4

Figure 4

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3.2.4. Depression

The meta-analysis of the relationship between catastrophizing and depression included 19 independent samples and resulted in a positive large effect size, with r = 0.49 (95% CI: 0.43-0.55, Table 4). The overall effect was significantly different from zero (z = 12.58, P < 0.01) and an additional 180 studies with null effects would be needed to reduce this to nonsignificance. Egger regression test of asymmetry resulted in a precision value = 0.49 (90% CI: −1.48 to 2.46, P = 0.61), indicating no significant amount of asymmetry, suggesting no significant amount of publication bias.23 The funnel plot indicates a roughly symmetric distribution of effect sizes (Fig. 5). The results of heterogeneity analyses (Q = 66.96, I2 = 73.12%) supported additional moderation analyses.

Figure 5

Figure 5

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3.2.5. Quality of life

The meta-analysis of the relationship between catastrophizing and QOL included 10 independent samples and resulted in a positive large effect size, with r = −0.48 (95% CI: −0.56 to −0.39, Table 4). The overall effect was significantly different from zero (z = 9.32, P < 0.01), with the fail-safe N analysis indicating that 91 additional studies with null effects would be needed to reduce the overall mean effect to nonsignificance. Egger regression test of asymmetry resulted in a precision value = 0.37 (90% CI: −2.10 to 2.84, P = 0.74), indicating no significant amount of asymmetry, suggesting no significant amount of publication bias.23 The funnel plot indicates a roughly symmetric distribution of effect sizes, further suggesting that publication bias is unlikely (Fig. 6). The Q-statistic of the overall effect was 30.31, with a substantial amount of heterogeneity (I2 = 70.31%), indicating the need for additional moderation analyses.

Figure 6

Figure 6

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3.3. Moderation

Moderator analyses for the relationship between catastrophizing, pain, and pain outcomes are shown in Tables 5–7. Among the continuous moderators and contrary to hypotheses, sex, age, pain intensity, and pain duration did not significantly moderate the relationship between catastrophizing and any of the pain outcomes. Similarly, among the categorical moderators, neither type of chronic pain nor catastrophizing measure significantly moderated (P > 0.05 for Q (b) values) any of the relationships between catastrophizing and pain outcomes. We could not conduct categorical moderation analyses of the relationship between catastrophizing and QOL due to having less than 2 studies for pain diagnosis or catastrophizing measure subgroups.

Table 5

Table 5

Table 6

Table 6

Table 7

Table 7

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3.4. Study quality

Study quality is reported in Table 2. The mean QR for studies was 5.97 (median = 6 and mode = 5). In line with previous studies,17,27,48,90 QR was examined as a moderator of subgroup differences in effect sizes. Studies were grouped using sample mode (5) as a cut point,90 with studies 6 or above coded as “higher quality” and studies scoring at or below 5 considered “lower quality.” Effect sizes between these 2 groups were compared. Study quality was not a significant moderator (P > 0.05 for Q (b) values) for any of the relationships between catastrophizing and pain outcomes. Detailed results are presented in Table 8.

Table 8

Table 8

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3.5. Subgroup analyses

Two study characteristics were explored to assess their impact on the mean effect size: (1) whether or not samples included any participants older than 18 years, and (2) whether or not samples contained any participants reporting pain duration less than 3 months. Mean effect sizes were estimated including and excluding studies with these characteristics (ie, participants older than 18 and participants with pain <3 months). As seen in Table 9, minimal differences in mean effect sizes were observed between the total study sample and the subgroups, indicating that these study characteristics did not substantially influence the results.

Table 9

Table 9

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4. Discussion

The current meta-analysis assessed the magnitude of the relationship between catastrophizing, pain, and functional outcomes in children with chronic pain. Effect sizes ranged from medium to large, with anxiety, depression, and QOL demonstrating strong associations with catastrophizing. Pain intensity and physical functioning had moderate associations with catastrophizing. These relationships were robust, minimizing the potential influence of publication bias and study quality. Sex, age, pain intensity rating, pain duration, pain diagnosis, and type of catastrophizing measure did not significantly moderate the relationship between catastrophizing and pain or functional/psychosocial outcomes.

Of the outcomes examined in the current meta-analysis, anxiety and depression had the strongest absolute relationships with catastrophizing. This strong relationship may be partially due to individuals having a general maladaptive thinking style that transcends pain-related stressors. Both in a broader psychological context and specifically within the chronic pain literature, catastrophizing has been characterized as a maladaptive thinking style in response to stress,6,24,104 frequently observed among individuals with anxiety12,102 and depressive disorders,32 as well as those with chronic pain.87 Thus, children who catastrophize in response to pain—and/or who appraise pain in catastrophic ways—may do similarly for nonpain stressors. This general cognitive–emotional style may explain the strong relationships between catastrophizing and anxiety and depression observed herein. Worth noting, a handful of studies point to conceptual overlap as a reason for the strong association between pain catastrophizing and negative mood (eg, depression and anxiety).1,46,105 Although we acknowledge these blurry demarcations, cross-sectional34,58,106 and longitudinal57 evidence supports the conceptual distinctiveness of pain catastrophizing. These conceptual issues are paralleled by concerns about measurement overlap and common-method variance. Subscale or item-level analysis between measures of catastrophizing, anxiety, and depression may clarify the relevance of these methodological issues. Unfortunately, because the majority of studies included in the current meta-analysis did not report subscale or item-level scores, we could not undertake such analyses ourselves.

The strong relationship between catastrophizing and emotional outcomes suggests that intervention efforts aimed at decreasing catastrophic thinking may reduce anxiety and depressive symptoms among children with pain. Only a few studies have examined this issue. Kashikar-Zuck et al.56 evaluated an 8-week CBT program for children with juvenile fibromyalgia and found that although catastrophizing decreased over the course of treatment, it did not mediate improvement in depressive symptoms. Similarly, Wicksell et al.125 found that catastrophizing did not mediate changes in depression in response to an acceptance and commitment therapy program (ranging from 7 to 20 therapy sessions) for pediatric chronic pain. Although these studies suggest that cognitive–behavioral and acceptance-based approaches can effectively reduce pain catastrophizing in children, much remains to be known about whether and how these changes translate into improvement in psychological functioning. Of note, these studies used less common measures of catastrophizing (PRI and PCQ), and neither assessed anxiety symptoms, thus constraining our understanding of these relationships.

Catastrophizing and physical functioning were moderately related in our analysis. The fear-avoidance model of chronic pain provides one lens through which to interpret this relationship.3,62,85,97 The fear-avoidance model posits that poor physical functioning results from pain-related catastrophizing and threat appraisals. Thinking about pain in this way increases fear of future pain, leading to avoidance of activities and ultimately to disuse and disability.97 This model has been applied to pediatric populations, lending support to the idea that intervening on pain catastrophizing could lead to improved physical functioning in children and adolescents with pain through decreases in their fear of pain and avoidance of activities. The biopsychomotor model offers another perspective on the connection between catastrophizing and physical functioning in children and adolescents. This model posits that pain behavior is an integral part of the pain system,103 serving communicative, protective, and social–relational purposes. Pain expression is a form of communication, transmitting information that has survival value while also soliciting attention and support for the pain sufferer.36,40,126 Although catastrophizing has been linked to increased communicative pain behaviors, specifically facial expressions of pain,72,118,119 the protective and social–relational aspects of the biopsychomotor model seem particularly relevant to the current findings regarding the link between catastrophizing and physical functioning. Children who endorse high levels of catastrophizing about their pain may, consequently, engage in protective behaviors (eg, guarding and bracing) that have been linked to increased functional disability.107 These behaviors may also trigger the social response system of proximal others, leading to solicitous behaviors from parents, teachers, and peers. Such solicitousness may reinforce the child's pain and avoidance behaviors, thereby leading to continued physical disuse and worse functional disability.

Quality of life was strongly associated with catastrophizing. In child and adolescent chronic pain samples, QOL is often measured with the PedsQL,18,52,64,71,72,74,76,112 which assesses physical, emotional, social, and school domains.115 Thus, the strong association between catastrophizing and QOL may be driven, in part, by the physical and emotional domains, which overlap with our other outcomes of interest (ie, anxiety, depression, and physical functioning) that demonstrated moderate–strong associations with catastrophizing. However, the PedsQL also assesses social and school functioning, domains of the pediatric pain experience that have received less attention in this literature. Children/adolescents with chronic pain frequently struggle with peer relations and academic achievement,31,82 and there is some research suggesting that catastrophizing contributes to poorer functioning in these domains.78 Pain expressions and behaviors might tie these intrapersonal and interpersonal factors together. As discussed above, although such behaviors may elicit emotional and functional support from others, they can also occasion more punitive responses, such as ignoring or expressions of anger or irritation,10 and are associated with interpersonal problems.61 Although the current findings support the notion that pain catastrophizing and general QOL are strongly related in children with pain, future research should take a more nuanced approach, separating the construct into individual domains to elucidate their unique associations with pain catastrophizing.

Catastrophizing and pain intensity were moderately associated. This relationship was the most modest and varied (r = −0.25 to 0.58) among those examined in our meta-analysis. Several physiological and neural explanations have been proposed to explain the link between catastrophizing and pain intensity; these include exaggerated muscle responses at the site of injury,86 altered hypothalamic–pituitary–adrenal axis activity,22,51 and lack of activation in brain regions responsible for top-down inhibitory control.92 Although these studies suggest that pain catastrophizing is related to greater activity in brain regions involved in affective processing of pain, attention to pain, and pain behaviors, all were conducted in samples of adults with chronic pain. Whether and how such explanations apply to children remains an open question. Erpelding et al.26 found that, among children with complex regional pain syndrome, pain catastrophizing was correlated with increased gray matter and activation in brain regions involved in motor function, sensorimotor integration, and anxiety-driven exacerbations of pain–these findings suggest that pain catastrophizing may predispose children and adolescents to develop chronic pain, experience greater pain intensity when they do have pain, and engage in particular behavioral and emotional responses to that pain. Additional studies are needed to better understand the extent to which catastrophizing is a cause and/or consequence of increased pain in children, as well as the neurophysiological underpinnings of these relationships.

The varying effect sizes–magnitude and direction–observed in the current meta-analysis suggest a complex relationship between catastrophizing and pain in children that is moderated by other factors. We examined several candidate moderators that were suggested by theory and previous studies; however, none significantly moderated the relationships examined herein. Although evidence suggests that the pain experience differs for boys and girls of varying ages,7,28 the samples included in our analyses were composed mostly of adolescent girls. In addition, because the majority (83%) of included samples reported moderate (Numeric Rating Scale = 4) to severe (Numeric Rating Scale = 7) average pain intensity, our moderation analyses of sex, age, and pain intensity may have lacked adequate power. Future, high-powered studies are needed to better understand whether and how these factors impact the relationship between catastrophizing and pain outcomes in children.

Pain duration and pain diagnosis were also not supported as moderators, which suggests that the catastrophizing–pain nexus is not altered by the length of time or the specific pain diagnosis a child/adolescent has been experiencing. However, it bears noting that the number of studies included in specific pain diagnoses subgroups was low (all Ns < 6), thus reducing confidence in the reliability of the null finding. Finally, pain catastrophizing measure did not explain significant heterogeneity in the observed relationships. Nevertheless, both Q and I2 values were substantially reduced within categorical moderation subgroups for catastrophizing measure and pain diagnosis, indicating a reduction of variability in effect size heterogeneity between studies.49

Findings from this meta-analysis have implications for treatment. Evidence supports the efficacy of several psychological interventions for reducing pain catastrophizing in pediatric samples–these include CBT for chronic pain,29,70 relaxation training,29 and acceptance and commitment therapy.29,124 These therapies often educate patients about pain catastrophizing, and some target self-reported catastrophic thoughts directly (ie, through cognitive restructuring) or indirectly (ie, through experiential exercises). Despite their wide use, much remains to be known about the effectiveness of such approaches–individually and/or collectively–as well their durability and mechanisms of action. Answers to these questions would provide insights for the further refinement of existing treatments and the formulation of new treatments to target catastrophic thinking and improve the functioning of children with pain. Results of the current meta-analysis suggest that such improvement may be especially achieved in the domains of anxiety and depression, as well as overall QOL.

Several limitations should be acknowledged. The majority of studies used clinical data, which introduces several forms of error, including selection bias and unsystematic administration of measures.93 In addition, clinical data sets are often used repeatedly for various publications, a detail that is not specifically and consistently disclosed, making it difficult to guarantee samples and corresponding effect sizes are unique. Furthermore, the variability in outcome measures used to assess a particular outcome (eg, physical functioning) may influence effect sizes. Another limitation, common in meta-analyses, is the file drawer problem.80 Although the results of fail-safe analyses instill confidence in the meta-analytic findings for all of our primary outcome variables, we cannot rule out the existence of unpublished studies that would have changed these findings had they been included. This meta-analysis only included a selection of outcomes that may be associated with catastrophizing. Unfortunately, other potentially important outcomes, such as school performance, are not commonly reported in the pediatric pain literature and thus were not included herein. Finally, all measures were self-reported and used similar item-response formats, thus, increasing the possibility that common method bias contributed to the observed associations.

Future studies should aim to elucidate possible moderators of the relationship between catastrophizing and pain outcomes because the current results indicate considerable between-study variability in the nature of this relationship. Future research may also investigate the effectiveness of current interventions in reducing catastrophic thinking and the magnitude of these effects over time. These findings would serve to enhance the individualization of treatments for chronic pain in children. The role of parents is important to examine as well. Many studies have reported significant relationships between parental catastrophizing and child pain outcomes,37,67,74 but these relationships are yet to be meta-analytically quantified.

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Conflict of interest statement

The authors have no conflict of interest to declare.

This research was partially supported by the National Institutes of Health under award number T32 AR055855. We confirm that there have been no closely related manuscripts that have been submitted for simultaneous consideration to this or another journal.

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References

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    Keywords:

    Meta-analysis; Catastrophizing; Child; Adolescent; Chronic pain; Pediatric; Functioning

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