Pain coping and catastrophizing in youth with and without cerebral palsy

Michael N. Vinkel; Gija Rackauskaite; John R. Østergaard; Nanna B. Finnerup; Mark P. Jensen

doi:10.1515/sjpain-2023-0062

Article Open Access

Pain coping and catastrophizing in youth with and without cerebral palsy

Michael N. Vinkel , Gija Rackauskaite , John R. Østergaard , Nanna B. Finnerup and Mark P. Jensen

Published/Copyright: March 7, 2024

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From the journal Scandinavian Journal of Pain Volume 24 Issue 1

Abstract

Objectives

The aim of this study is to compare the use of pain coping strategies and pain catastrophizing in youth with and without cerebral palsy (CP), and to examine how these two groups differ with respect to the associations between pain coping, catastrophizing, and measures of psychological function and sleep disturbance.

Methods

Twenty-seven individuals with CP and 49 healthy controls aged 15–22 were included in this cross-sectional observational study. Pain was assessed using a semi-structured interviews and participants completed measures of pain coping, pain catastrophizing, psychological function, and sleep.

Results

Youth with CP used information seeking and problem solving (p = 0.003, Cohen’s d (d) = −0.80) and sought social support (p = 0.044, d = −0.51) less often, and used internalizing as a coping strategy more often (p = 0.045, d = 0.59) than healthy controls. The use of information seeking and problem solving correlated more strongly with measures of depression (p = 0.023, Cohen’s f (f) = 0.08) and sleep disturbance (p = 0.022, f = 0.08), while behavioral distraction correlated more strongly with measures of anxiety (p = 0.006, f = 0.11) and sleep disturbance (p = 0.017, f = 0.09) in youth with CP, compared to healthy controls.

Conclusions

The study findings raise the possibility that youth with CP may benefit more in terms of psychological function and sleep quality from coping training interventions that focus on behavioral distraction, information seeking, and problem solving. Research to test these ideas in additional samples of youth with CP is warranted.

Keywords: cerebral palsy; pain; coping; catastrophizing; psychological function

1 Introduction

Chronic pain is a common comorbidity of cerebral palsy (CP). The estimated prevalence of pain in children and youth with CP ranges from 27 to 77%, depending on age, ambulation, and how pain is defined and measured [1–7]. Increased muscle tone, contractures, and misalignment of joints are common causes of musculoskeletal pain affecting extremities and the back [8]. Other types of pain such as headache and visceral pain (e.g., abdominal pain from constipation or gastro-esophageal reflux) also have a high prevalence [1,9,10]. Additionally, many individuals with CP experience acute pain when they undergo medical procedures, such as surgery and botulinum toxin injections. Furthermore, a cornerstone in the management of CP is regular physiotherapy, where assisted stretching and range of motion manipulation are often reported as painful [11,12]. Consequently, not all painful experiences are entirely treatable nor preventable, and the prevalence of pain remains high in individuals with CP, even when the person is being followed closely by a multidisciplinary team of healthcare professionals.

Pain is known to have a negative impact on different quality of life domains – such as daily activities, physical activity, and sleep quality [13–19] – in individuals with CP. Studies also show that pain is associated with mental health problems in children with CP [20]. Symptoms of depression and anxiety are also more common in people with CP than in healthy controls [20–25], perhaps related in part to the increased risk of pain in this population.

Coping refers to the efforts made by people to manage stressors, including stressors associated with pain [26]. Some pain coping strategies are adaptive and tend to be associated positively with better physical and psychological function, while other strategies are maladaptive and tend to be positively associated worse function [27]. Whether a coping strategy is adaptive or maladaptive depends on the situation and context. Active coping strategies – such as information seeking and problem solving – are generally considered adaptive, whereas passive strategies – such as avoidance and pain-contingent rest – are generally considered maladaptive [28]. Studies support a positive association between maladaptive pain coping and symptoms of depression in adults with CP [29,30]. In this article, we will use the term “function” as a broad term covering psychological function (e.g., depression and anxiety symptom severity) and sleep quality (e.g., severity of sleep disturbance).

Pain catastrophizing is often viewed as a maladaptive coping strategy, because it is reliably associated with worse function [31]. Pain catastrophizing has been defined as “an exaggerated negative mental set brought to bear during painful experiences” [32]. Ultimately, how individuals with CP cope with their pain may influence their function.

The pain coping strategies that individuals use are influenced by numerous factors, including past experiences with pain. Thus, people who have chronic pain will develop strategies to manage that pain, which may differ from the pain coping strategies used by individuals who do not have chronic pain. For example, Thastum and colleagues found less use of behavioral distraction in children with juvenile idiopathic arthritis compared to healthy controls [33].

To our knowledge, only one study has compared coping strategies used by children and youth with CP with those used by healthy controls [34]. These investigators found that children and youth with CP used cognitive self-instruction, distraction, and problem solving less than typically developed children and adolescents. However, the proportion of males and females in the two groups was significantly different [35], and this sex difference could have explained the significant effects found. Moreover, this study did not examine how different coping strategies were associated with function in the two groups. If coping responses are found to have the same association with function across different groups (e.g., healthy controls vs those with CP), then this would mean that interventions that teach and encourage the coping responses found to be most adaptive should be similarly effective across different pupations of individuals with pain. On the other hand, if the associations between coping and function differ across different populations, this would suggest that interventions would need to be developed that are specific to different populations (i.e., interventions to teach and encourage coping responses specifically important to that population).

Given these considerations, here we sought to address knowledge gaps regarding how youth with CP and healthy controls may be similar or different with respect to (1) pain catastrophizing levels and pain coping strategies used and (2) the associations between catastrophizing, pain coping, and function. Based on the findings from prior research, cited previously, we hypothesized that youth with CP would use fewer active pain coping strategies than healthy controls. Regarding the second study aim, we hypothesized that pain coping and catastrophizing would be significantly associated with function, but we did not have any a priori hypotheses regarding how the associations between pain coping, catastrophizing, and function would differ between youth with CP and healthy controls. Thus, we considered tests related to the second study aim to be exploratory.

2 Methods

2.1 Participants

A total of 27 individuals with CP and 49 age- and sex-matched controls were enrolled in this cross-sectional, observational study. The individuals with CP were recruited though pediatric departments in the Central Denmark Region and had to meet the following eligibility criteria: (1) CP with GMFCS level I–III, (2) no significant chronic diseases not associated with CP, (3) age 15–22 years, (4) normal IQ, (5) ability to speak and understand Danish, and (6) no family history of hereditary neurological diseases. The age range for study inclusion was chosen to help control for potential sample heterogeneity due to age-related changes that occur in the causes of CP-related pain as well as in coping strategies as people age from childhood into adolescence, while also allowing for a sample that represents a young population [34,36]. Healthy controls were recruited using Aarhus University Hospital’s official social media channels. Controls were required to not have any significant chronic diseases (including no primary pain syndromes as defined by the International Classification of Diseases 11th Revision), and fulfill criteria (3)–(6). We sent invitation letters directly to individuals with CP age 18–22 years, while letters were sent to parents of participant’s age 15–17 years.

All participants were examined at the Danish Pain Research Center, Aarhus, Denmark. We performed a semi-structured interview assessing demographics, pain, and CP. Moreover, participants completed a series of questionnaires assessing pain coping, pain catastrophizing, and function.

2.2 Semi-structured interview

We assessed pain using a predetermined open question “Do you have any persistent or recurrent pain problem?” If the answer was “No,” we presented examples of common recurrent pain problems in youth. “Some have a tendency of muscle or joint pain following physical activity, while some have recurrent headache, stomachache, or back pain. Is this something you are familiar with?” Both to insure that no pain problem was neglected and to confirm eligibility criteria of the healthy controls. Educational level was assessed and physical activity was computed from the International Physical Activity Questionnaire (IPAQ) [37]. Furthermore, participants with CP were asked about previous CP-related surgery, physiotherapy, and other treatments (botulinum toxin injections).

2.3 Pain coping

The Danish version of the 39-item Pain Coping Questionnaire (PCQ) has been validated in children and youth aged 8–17 years [33]. Despite being developed for children, research suggest that the questionnaire is also reliable in adults [38], and it has previously been used in young adults [39]. Respondents to the PCQ are asked to rate how often they use different pain coping strategies when they are in pain using a 5-point Likert scale (ranging from “Never” to “Very often”). The PCQ items are scored into seven subscales: Information Seeking and Problem Solving, Cognitive Distraction, Behavioral Distraction, Positive Self-Statement, Seeking Social Support, Internalizing, and Eternalizing. One of the items elicits information on whether the participant asks a doctor or nurse for help when in pain. However, youth with CP are usually in regular contact with their physiotherapist. With permission from the author of the PCQ, we changed the question about help seeking to include physiotherapists. Higher scores on each scale reflect higher levels of the coping responses assessed.

2.4 Pain catastrophizing

The 13-item Pain Catastrophizing Scale (PCS) is designed to measure three domains of catastrophizing (rumination, magnification, and helplessness), as well as overall catastrophizing. Respondents to the PCS are asked to indicate the degree to which or frequency with which they respond to pain with a specific catastrophizing response on a 5-point Likert scale (from 0 = “Not at all” to 4 = “All the time”). The PCS was initially validated in adults [40] and later a version was validated in children aged 8–16 [41]. The age group in this study crosses the line from child to adult, which poses a challenge when choosing a questionnaire. For the current study, we chose the adult version of the PCS, as a Danish version already existed which had shown high reliability [42].

2.5 Psychological function and sleep disturbance

Psychological function and sleep disturbance were assessed using three different Patient-Reported Outcomes Measurement Information System (PROMIS) short forms: The Anxiety Short Form 6a v.1.0, Depression Short Form 6a v1.0, and Sleep Disturbance Short Form 6a v1.0. The short forms are available in Danish, consist of six items each, and provide respondents with 5-point Likert scale response options. The Anxiety and Depression Short Forms assess (from 1 = “Never” to 5 = “Always”) how often participants experience different feelings and symptoms during the past 7 days. The first item of the Sleep Disturbance Short Form assesses quality of sleep during the past 7 days (from 5 = “Very poor” to 1 = “Very good”), while the second item assesses if sleep was refreshing (from 5 = Not at all” to 1 = “Very much”). The four last items in the Sleep Disturbance Short Form assess how often participants had different sleep-related problems in the past 7 days (from 1 = “Never” to 5 = “Always”). PROMIS measures use a t-score metric with a mean of 50 and a standard deviation of 10. The total raw score was translated into a T-score for each participant using PROMIS scoring manuals. Higher PROMIS scores indicate more of the domain assessed, as reflected by the scale score label (i.e., more depression, anxiety, or sleep disturbance, in this case).

2.6 Data analysis

We first assessed the normality of the distributions of the study variables using the Shapiro–Wilk test, supported by graphical interpretation of histograms and Q–Q plots. The Shapiro–Wilk test indicated that most PCQ and PCS subscales were approximately normally distributed, while PROMIS scores assumed a more skewed distribution. Regarding missing values, one participant was missing a single item from the PCQ Cognitive Distraction subscale, and this same participant was missing an item from the PCS Helplessness subscale. Another participant had a single missing item in the PCQ Positive Self-Statement subscale. For these participants, pro-rated subscale scores were computed using an average of the items responded to. The guidelines for IPAQ scoring are that the respondents must provide a response to all items (i.e., anything other than “I don’t know”) in order for the scale to be scored. Because of this, six healthy controls and one participant with CP were not included in the analysis involving the IPAQ scale.

For descriptive purposes, we computed the frequencies and percentages (for categorical variables) and mean and standard deviation (for continuous variables) of the sociodemographic and study variables. To account for unequal variance and unequal sample sizes, differences between groups on the descriptive variables were tested using Welch’s t-test (which is robust under skewed distribution) for continuous variables, while the chi-square test was used for categorical variables. IPAQ scores assessing physical activity were computed as median Metabolic Equivalent Task (MET) minutes per week.

To address the first study aim, examining hypothesized differences between the CP group and the healthy control group with respect to pain coping and catastrophizing responses, we conducted a series of 11 Welch’s t-tests, using the PCS and PCQ scale scores as dependent variables. Effect sizes for the t-tests were computed as Cohen’s d (i.e., Cohen’s d = 0.2 indicates a small effect size, Cohen’s d = 0.5 indicates a medium effects size, and Cohen’s d = 0.8 indicates a large effect size).

For descriptive purposes, before addressing the second study aim, we computed two series (one for each group) of 33 zero-order correlations (one for each measure of pain coping and catastrophizing as the independent variable), with measures of depression, anxiety, and sleep disturbance as the dependent variable. Effect sizes are reported as Spearman’s rho (i.e., rho = 0.2 indicates a small effect size, rho = 0.4 indicates a medium effects size, and rho = 0.6 indicates a large effect size).

Finally, to address the second study aim, we conducted a series of 33 regression analyses (one for each measure of coping and catastrophizing), with measures of depression, anxiety, and sleep disturbance as the criterion variable. We entered group (dummy coded as CP vs healthy control) and pain coping and catastrophizing subscale scores together in step 1, and their interaction in step 2. The second step tests whether group moderates the effect of coping or catastrophizing on measures of function. Effect sizes for the R ²-change were computed as Cohen’s f (i.e., Cohen’s f = 0.02 indicates a small effect size, Cohen’s f = 0.15 indicates a medium effects size, and Cohen’s f = 0.35 indicates a large effect size). Given the low sample size in the current study, we evaluated the study aims with respect to the effect sizes over statistical significance, although we planned to report both. All statistical analyses were conducted using Stata (v. 15.1, Statacorp LLC, College Station, TX, USA).

Due to European and Danish law protecting patient privacy, there may be restrictions for obtaining individual data and a request needs to be sent to the corresponding author.

3 Results

3.1 Demographics

The demographic characteristics of the two groups are presented in Table 1. Twenty-seven individuals with CP and 49 healthy controls participated in the study. There were no statistically significant differences in age (p = 0.63) or sex (p = 0.81) between the two groups. The prevalence of persistent or recurrent pain was significantly higher in youth with CP 70% (CI: 50–85) than in healthy controls 14% (CI: 7–27). When presented with examples of pain no additional pain problems appeared in participants answering “no” to the gate question on pain.

Table 1

Sociodemographic characteristics and study variables of youth with cerebral palsy and healthy controls

	Cerebral palsy (n = 27)	Healthy controls (n = 49)	Group differences
	Mean (SD) or N (%)	Mean (SD) or N (%)	p-value
Age – mean (SD)	19.5 (2.6)	19.7 (2.2)	0.633
Sex
Male	14 (51%)	24 (49%)	0.811
Female	13 (48%)	25 (51%)
Pain
Recurrent or chronic pain	19 (70%)	7 (14%)	<0.001
Pain during the past week	19 (70%)	5 (10%)	<0.001
GMFCS
Level I	12 (44%)
Level II	15 (56%)
CP type
Spastic unilateral	21 (78%)
Spastic bilateral	5 (19%)
Mixed	1 (4%)
CP specific
Physiotherapy the past year	15 (56%)
Previous CP-related surgery	17 (63%)
Previous botulinum toxin injections	16 (59%)
Psychological function (PROMIS)
Depression, T-score	47.5 (9.3)	45.4 (6.9)	0.307
Anxiety, T-score	52.2 (9.1)	50.7 (7.0)	0.461
Sleep disturbance, T-score	51.0 (11.1)	49.4 (6.3)	0.492
Physical activity (IPAQ)
MET-minutes/week	1,555 (1,488)	2,763 (2,506)	0.029
Education
Current education for students
Primary school	5 (19%)	4 (8%)
High school	6 (22%)	1 (2%)
Postsecondary education	6 (22%)	18 (37%)
University	2 (7%)	13 (27%)
Completed education for non-students
Primary school	2 (7%)	1 (2%)
High school	6 (22%)	12 (24%)

Data are n (%) for categorical variables and mean (SD) for continuous variables. GMFCS = gross motor function classification system; CP = cerebral palsy, PROMIS = patient-reported outcomes measurement, IPAQ = International Physical Activity Questionnaire, MET = Metabolic Equivalent Task.

3.2 Coping and catastrophizing

Table 2 compares the mean PCQ and PCS scale scores of youth with CP and healthy controls. Youth with CP had significantly lower scores in the Information Seeking and Problem Solving (p = 0.003, d = −0.80, large effect) and the Seeking Social Support (p = 0.044, d = −0.51, medium effect) subscales compared to healthy controls. Conversely, youth with CP scored higher in the Internalizing subscale compared to healthy controls (p = 0.045, d = 0.59, medium effect). There were no significant group differences in the other PCQ or any of the PCS scores, and all of the effect sizes for these differences were small (d’s ranging from 0.01 to 0.32).

Table 2

Comparison of pain coping and pain catastrophizing in youth with and without cerebral palsy

	Cerebral palsy (n = 27)		Healthy controls (n = 49)		Group differences
	Mean (SD)	Median	Mean (SD)	Median	Welch’s t-test (t)	p	Cohen’s d
Pain coping (PCQ)
Information seeking and problem solving	2.6 (0.8)	2.6	3.1 (0.7)	3.1	−3.19	0.003	−0.80
Cognitive distraction	3.0 (0.9)	3.0	3.1 (0.7)	3.0	−0.64	0.523	−0.17
Behavioral distraction	3.1 (0.8)	3.0	3.3 (0.8)	3.2	−1.08	0.285	−0.26
Positive self-statement	3.2 (0.8)	3.2	3.4 (0.6)	3.4	−0.84	0.403	−0.22
Seeking social support	2.6 (1.0)	2.4	3.0 (0.9)	3.0	−2.07	0.044	−0.51
Internalizing	2.3 (0.9)	2.2	1.9 (0.5)	1.8	2.08	0.045	0.59
Externalizing	1.5 (0.7)	1.4	1.5 (0.6)	1.2	−0.05	0.960	−0.01
Pain catastrophizing (PCS)
Total catastrophizing	15.5 (9.5)	16	13.4 (8.1)	12	0.98	0.330	0.24
Rumination	6.6 (4.3)	8	5.9 (3.4)	6	0.69	0.496	0.18
Magnification	2.8 (2.7)	2	2.6 (1,9)	3	0.24	0.806	0.07
Helplessness	6.1 (4.4)	5	4.9 (4.0)	4	1.31	0.196	0.32

PCQ = Pain Coping Questionnaire; PCS = Pain Catastrophizing Scale. Effect size (Cohen’s d) values that are medium or larger in size (i.e., ≥0.50) are presented in bold face.

3.3 Function

Table 3 shows, for descriptive purposes, the Spearmen’s rho correlation coefficients between pain coping and function, and pain catastrophizing and function, for both groups. Information Seeking and Problem Solving correlated with depression scores with a medium effect size (rho = 0.40, p = 0.040) in youth with CP, while this correlation evidenced a non-significant trend for the anxiety scores with a small to medium effect size (rho = 0.34, p = 0.086). In both the CP and control group, we found medium effect sizes for the associations between Externalizing and depression scores (rho’s = 0.38 and 0.39, p’s = 0.050 and = 0.005) and Internalizing and depression scores (rho’s = 0.37 and 0.39, p’s = 0.055 and = 0.005). The effect sizes were medium or larger in the CP group with regard to the association between Externalizing and anxiety scores (rho = 0.51, p = 0.007) and Internalizing and anxiety scores (rho = 0.46, p = 0.016). Furthermore, Internalizing correlated with sleep disturbance scores with a medium effect size in youth with CP (rho = 0.42, p = 0.030). Considering pain catastrophizing, we found medium effect sizes of the correlation between Helplessness and depression scores (rho = 0.44, p = 0.023), Magnification and anxiety scores (rho = 0.46, p = 0.015), and Helplessness and sleep disturbance scores (rho = 0.50, p = 0.008) in the CP group.

Table 3

Spearman’s rank order correlation coefficients between measures of coping, catastrophizing, and four measures of function in patients with cerebral palsy and healthy controls

	Depression (PROMIS)		Anxiety (PROMIS)		Sleep disturbance (PROMIS)
	CP group	Ctrl. group	CP group	Ctrl. group	CP group	Ctrl. group
Pain coping (PCQ)
Information seeking and problem solving	0.40*	−0.14	0.34	−0.11	0.33	−0.25
Cognitive distraction	0.04	0.02	0.11	0.01	−0.08	0.04
Behavioral distraction	0.06	−0.00	0.34	−0.11	0.28	−0.14
Positive self-statement	0.18	0.00	0.23	0.09	0.16	0.14
Seeking social support	−0.00	−0.01	0.16	0.00	0.02	−0.16
Internalizing	0.37	0.39**	0.46*	0.29*	0.42*	−0.13
Externalizing	0.38*	0.39**	0.51**	0.16	0.14	0.11
Pain catastrophizing (PCS)
Total catastrophizing	0.38	0.28	0.42*	0.27	0.37	0.03
Rumination	0.27	0.17	0.34	0.21	0.34	0.02
Magnification	0.31	0.21	0.46*	0.29*	0.10	0.16
Helplessness	0.43*	0.30*	0.36	0.24	0.50**	0.01

*p < 0.05; **p < 0.01. CP = cerebral palsy; Ctrl. = control; PCQ = Pain Coping Questionnaire; PCS = Pain Catastrophizing Scale; PROMIS = patient-reported outcomes measurement.

The findings from analyses to address the second study aim are presented in Tables 4–6. The correlation between Information Seeking and Problem Solving and depression score was significantly stronger in youth with CP than in healthy controls (p = 0.023) with a small to medium effect size (Cohen’s f = 0.08). There were no other significant group differences in the strength of correlation between pain coping and catastrophizing and measures of depression and all of the effect sizes for these differences were small (f’s ranging from 0.01 to 0.04). Furthermore, the correlation between Behavioral Distraction and anxiety score was stronger in youth with CP (p = 0.006) with a small to medium effect size (Cohen’s f = 0.11). The other effect sizes were small (f’s ranging from 0.01 to 0.05) with regard to the difference in strength of correlation between pain coping and catastrophizing and measures of anxiety in the two groups. Finally, two coping mechanisms, Behavioral Distraction (p = 0.017, f = 0.09) and Information Seeking and Problem Solving (p = 0.022, f = 0.08) correlated stronger (with small to medium effect sizes) with sleep disturbance scores in youth with CP than in healthy controls.

Table 4

Hierarchical regression analysis for variables predicting depression

	Step 1 R ²: group and predictor	Step 2 R ²-change: group × predictor interaction	F change	p	Cohen’s f²
Pain coping (PCQ)
Information seeking and problem solving	0.03	0.07	5.39	0.023	0.08
Cognitive distraction	0.02	<0.01	0.04	0.852	0.01
Behavioral distraction	0.02	<0.01	0.21	0.649	0.01
Positive self-statement	0.03	<0.01	0.41	0.525	0.01
Seeking social support	0.02	<0.01	0.03	0.867	0.01
Internalizing	0.14	<0.01	0.43	0.516	0.01
Externalizing	0.19	0.01	0.93	0.338	0.01
Pain catastrophizing (PCS)
Total catastrophizing	0.11	0.01	0.88	0.352	0.01
Rumination	0.07	<0.01	0.65	0.423	0.01
Magnification	0.06	<0.01	0.06	0.815	0.01
Helplessness	0.12	0.02	1.38	0.244	0.02

PCQ = Pain Coping Questionnaire; PCS = Pain Catastrophizing Scale. Effect size (Cohen’s f ²) values of 0.02 indicate a small effect size, 0.15 a medium effects size, and 0.35 a large effect size.

Bold values denote a p-value less than 0.05.

Table 5

Hierarchical regression analysis for variables predicting anxiety

	Step 1 R ²: group and predictor	Step 2 R ² change: group × predictor interaction	F change	p	Cohen’s f²
Pain coping (PCQ)
Information seeking and problemsolving	0.02	0.05	3.51	0.065	0.05
Cognitive distraction	0.02	<0.01	0.23	0.631	0.01
Behavioral distraction	0.02	0.10	7.92	0.006	0.11
Positive self-statement	0.06	<0.01	0.34	0.563	0.01
Seeking social support	0.03	0.01	1.06	0.307	0.01
Internalizing	0.14	<0.01	0.04	0.847	0.01
Externalizing	0.04	<0.01	0.72	0.399	0.01
Pain catastrophizing (PCS)
Total catastrophizing	0.13	0.01	0.90	0.346	0.01
Rumination	0.08	<0.01	0.58	0.449	0.01
Magnification	0.15	<0.01	0.29	0.590	0.01
Helplessness	0.09	<0.01	0.33	0.567	0.01

PCQ = Pain Coping Questionnaire; PCS = Pain Catastrophizing Scale. Effect size (Cohen’s f ²) values of 0.02 indicate a small effect size, 0.15 a medium effects size, and 0.35 a large effect size.

Bold values denote a p-value less than 0.05.

Table 6

Hierarchical regression analysis for variables predicting sleep disturbance

	Step 1 R ²: group and predictor	Step 2 R ² change: group × predictor interaction	F change	p	Cohen’s f²
Pain coping (PCQ)
Information seeking and problem solving	0.01	0.07	5.45	0.022	0.08
Cognitive distraction	0.01	<0.01	0.18	0.673	0.01
Behavioral distraction	0.02	0.08	5.97	0.017	0.09
Positive self-statement	0.02	<0.01	0.40	0.530	0.01
Seeking social support	0.01	0.03	2.21	0.142	0.03
Internalizing	0.03	0.04	3.31	0.073	0.04
Externalizing	0.02	0.02	1.37	0.246	0.02
Pain catastrophizing (PCS)
Total catastrophizing	0.05	0.03	2.63	0.109	0.03
Rumination	0.05	0.04	3.18	0.079	0.04
Magnification	0.01	<0.01	0.31	0.580	0.01
Helplessness	0.06	0.05	4.43	0.039	0.05

PCQ = Pain Coping Questionnaire; PCS = Pain Catastrophizing Scale. Effect size (Cohen’s f ²) values of 0.02 indicate a small effect size, 0.15 a medium effects size, and 0.35 a large effect size.

Bold values denote a p-value less than 0.05.

4 Discussion

Concerning the first study aim, we found that youth with CP used Information Seeking and Problem Solving less often than healthy controls. This finding is consistent with that of Chaleat-Valayer and colleagues measuring coping using the Pediatric Pain Coping Inventory (PPCI) [34]. On the other hand, and inconsistent with the findings from Chaleat-Valayer and colleagues, we found a significant (and large effect size) difference in how often youth with CP vs healthy controls seek social support.

The significant effects regarding information seeking and problem solving may be related to that fact that youth with CP in Denmark have regular contact with healthcare professionals and they have “disease educated” parents. Consequently, people with CP (at least in the age group studied here) may have the access to individuals who are able to provide them with information about, and assist them with, disease-related problems. This access may make it easier for individuals with CP than healthy controls to seek information and be able to problem solve.

A possible explanation for the discrepant finding in our study vs the study by Chaleat-Valayer and colleagues regarding how often youth with CP vs healthy controls seek social support might be the phrasing of the questions in the PCQ (used in this study) compared to the PPCI (used by Chaleat-Valayer and colleagues). While questions in the PCQ assessing the Seeking Social Support subscale put emphasis on “feelings” (e.g., “When I am hurt or in pain for a few hours or days, I talk to a friend about how I feel”), questions in the PPCI focus more on the activity of seeking social contact (e.g., “When I feel pain, I visit a friend”). Consequently, there is a difference in the kind of support sought and the means of seeking this kind of support in the two questionnaires. Neither the PPCI nor the PCQ examines whether respondents receive social support or not; only if they seek that support. As a result, the actual receipt of social support may be independent of the person’s own coping behavior. In addition, more recently, social media has enabled people with the same disabilities to easily connect with each other and their friends. Consequently, cultural differences in usage of social media platforms and the availability of online forums might affect how social support is sought.

Prior to addressing the second study aim, we examined simple correlations between different pain coping and catastrophizing strategies and measures of function to identify adaptive and maladaptive patterns. Internalizing and externalizing emerged as maladaptive coping strategies correlating with more frequent symptoms of depression in both groups and anxiety in the CP group. Furthermore, in both groups, pain catastrophizing patterns like magnifying the pain problem and helplessness correlated with the presence of symptoms of anxiety and depression, respectively. These results are in line with those from previous studies showing a link between catastrophizing and psychological function in adults with CP [30,31]. Our second study aim builds on these findings by examining the group differences of these associations.

We found no medium or large effect size differences in the strength of correlations between pain coping and catastrophizing and measures of psychological function. However, the correlation between Information Seeking and Problem Solving and depression score, and the correlation between Behavioral Distraction and anxiety score were significantly stronger in the CP group, with small to medium effect sizes. These results indicate that while catastrophizing may be associated with psychological function in general, the ability (or lack thereof) of seeking information, solving one’s own problem, and behavioral distraction appear to be more important to youth with CP than to healthy controls with regard to function. The correlation between Information Seeking and Problem Solving and sleep disturbance score, and the correlation between Behavioral Distraction and sleep disturbance score were significantly stronger in the CP group, with small to medium effect sizes. Although it is not possible to draw causal conclusions from the cross-sectional data in this study, the findings do suggest the possibility that there may be modifiable factors that could impact psychological function and sleep quality in youth with CP. If the results here are found to be reliable and future research shows the coping responses here as key causal factors of function, then it would be important to include a focus on increasing information seeking, problem solving, and behavioral distraction skill training when designing interventions for pain management for children with CP. Research to evaluate these possibilities is warranted.

The study has a number of limitations that should be considered when interpreting the results. Of primary importance is the low sample size, which was determined by the number of youth with CP willing to participate during the study period. This reduces power and can result in unreliable statistics, and therefore limits our ability to detect significant effects or place a great deal of trust in the reliability of the findings. Additionally, the number of regression analyses increases the risk of type 1 errors. Although the prevalence of pain in CP is high, the prevalence of CP is very small, with only 2/1,000 children having this diagnosis [43]. Furthermore, the prevalence of cognitive impairment in children with CP is estimated to 30–40% and many individuals with CP are not able to participate in studies using self-reported outcome measures [44]. As a result, recruiting large samples of children with CP is very challenging. Still, the topic of pain coping is worth examining even with a smaller sample size, because the findings can be used to guide future research, and can also be used as a benchmark for comparing the findings from future studies. If the same effects emerge in a series of several studies, each of which has a small sample size, this substantially increases the potential reliability of those findings. This is one of the reasons we focused on effect sizes over significance levels when interpreting the findings. A second limitation is that the study included a very select group of youth with CP. Thus, the findings may not generalize to the population of children with CP. Similarly, we excluded from the healthy control group children with primary pain conditions such as migraine. Therefore, the prevalence of pain in our control condition (14%) is lower than that found in the general population, where around 20% (CI: 18–21) are having almost daily pain [45,46]. The reason for excluding healthy controls with migraine and not those with recurring knee pain, was to ensure that controls were in fact healthy, but still represented a general population where recurring pain is a normal phenomenon. Finally, we found a group difference in MET-minutes per week, meaning that the control group used more energy per week on physical activity compared to the CP group. Additionally, pain was much more prevalent in the CP group. One explanation may be that exercise reduces the severity of chronic pain. However, the quality of the evidence examining the effect of physical activity on chronic pain is low [47]. It is more likely that people with CP are less active because of chronic pain, motor disabilities, or a combination thereof.

5 Conclusions

Despite the study’s limitations, the findings provide new and important information regarding the role that coping and catastrophizing play as predictors of function in children with CP, relative to a sample of otherwise healthy children. The differences found suggest the possibility that having CP, or having CP-related pain, may have an impact on how children think about and cope with pain, and these differences should be taken into account when designing intervention strategies to test in this population. Specifically, the findings suggest that youth with CP seek social support and use information seeking and problem solving to a lesser extent than healthy controls, and the youth with CP may possibly benefit most in terms of function from coping training interventions that focus on behavioral distraction, information seeking, and problem solving coping strategies. Research to test these ideas is warranted.

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Acknowledgments

The authors sincerely thank study nurses Bente Christensen and Rud Bugge Sørensen for their assistance and Niels Trolle Andersen for his support on statistical analyses.

Research ethics: The study complied with all relevant national regulations, institutional policies and is in accordance with the tenets of the Helsinki Declaration (as amended in 2013), and has been approved by the Central Denmark Region Committees on health Research Ethics (1-10-72-349-18) and registered at the Danish Data Protection Agency (1-16-02-343-18).
Informed consent: Prior to data collection, all participants (or legal guardians of participants under 18 years) provided written informed consent.
Author contributions: All authors have accepted responsibility for the entire content of this manuscript and approved its submission.
Competing interests: Nanna B. Finnerup is a Section Editor of the Scandinavian Journal of Pain. Authors declare no conflicts of interest in relation to this study. Outside the submitted work, N.B.F. has acted as a paid consultant to Almirall, NeuroPN, Vertex, Nanobiotix, and Novartis Pharma, has done consultancy work for Biogen, Merz, and Confo Therapeeutics for Aarhus University, and has received funding for research from IMI PainCare, an EU IMI 2 (Innovative medicines initiative) public–private consortium and the companies involved are: Grunenthal, Bayer, Eli Lilly, Esteve, and Teva.
Research funding: This study was supported by the Elsass Foundation, grant number 18-3-0096.
Data availability: The datasets generated during and/or analyzed during the current study are available from the corresponding author on reasonable request. Due to European and Danish law protecting patient privacy, there may be restrictions for obtaining individual data.

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Received: 2023-05-22

Revised: 2023-11-15

Accepted: 2023-12-08

Published Online: 2024-03-07

This work is licensed under the Creative Commons Attribution 4.0 International License.

Articles in the same Issue

https://doi.org/10.1515/sjpain-2023-0062

Keywords for this article

cerebral palsy; pain; coping; catastrophizing; psychological function

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