TY - JOUR
AU - Holmbeck, Grayson N
AB - Abstract Objective This study examined: (a) the nature and prevalence of pain in youth with spina bifida (SB) (b) common coping responses to pain, and (c) longitudinal, bi-directional associations between internalizing symptoms and pain characteristics. Methods Data were collected from youth (N = 140, 53.6% female, ages 8–15 at Time 1) and their parents and teachers at two time points spaced 2 years apart. Youth reported on several pain characteristics and coping responses. Multiple informants reported on child internalizing symptoms. Evaluation of Aims 1–3 was based on descriptive analyses, bivariate correlations, and linear and logistic regressions. Results About 25% of the sample reported chronic pain (e.g., experiencing pain one or more times per week over the past 3 months) at Time 1 or 2, with roughly one-third of this chronic pain subsample reporting chronic pain both time points. Pain was usually rated as mild in intensity for the full sample and most commonly experienced in the head, abdomen, and back, and described as “aching.” Youth with chronic pain reported significantly higher pain intensity and tended to use condition-specific methods to cope with pain (e.g., taking off braces). In 2 of 10 analyses, internalizing symptoms at Time 1 were associated with chronic pain and pain intensity at Time 2. Conclusions Roughly one-fourth of youth with SB are at risk for experiencing chronic pain, highlighting the need for increased assessment and treatment of pain in this population. Youth psychological functioning appears to more often precede, rather than being a consequence of pain symptoms. anxiety, chronic pain, coping, depression, pain, spina bifida Introduction Spina bifida (SB) is a relatively common congenital birth defect that occurs in roughly 3 out of every 10,000 births in the United States (National Birth Defects Prevention Network, 2010). SB is associated with a number of complications including bowel and bladder incontinence, varying degrees of paralysis of the lower extremities, orthopedic conditions, hydrocephalus, and neurocognitive issues, all of which require youth to follow a demanding medical regimen (Zukerman, Devine, & Holmbeck, 2011). Due to the medical sequelae related to this condition, youth with SB are also at risk for secondary physical conditions, including chronic pain (Clancy, McGrath, & Oddson, 2007; Oddson, Clancy, & McGrath, 2006). The negative impact of chronic pain on the physical and psychosocial well-being of children and their families is well-established (Liossi & Howard, 2016); nevertheless, research investigating the nature, prevalence, and development of pain in youth with SB remains limited. Of the few studies conducted to date, research suggests that pain is both prevalent and impactful in this population. One study of children and adolescents with SB (ages 8–19 years) found that over half of their sample experienced pain at least once per week (Clancy et al., 2007). Moreover, youth with SB may be at risk for untreated pain because parents often underestimate their child’s pain (Clancy et al., 2007). Prior research has begun to report on the type and location of pain in youth with SB, the etiology of which may in part be driven by extensive medical sequelae related to this condition. This research has found that youth with SB most commonly experience headache due to shunt infection or malfunction (Stellman-Ward, Bannister, Lewis, & Shaw, 1997), and musculoskeletal pain and joint pain due to ambulation method (i.e. overuse of certain muscles necessary for using wheelchairs or crutches; Marge, 1994). However, previous studies have not examined pain characteristics longitudinally wherein one could identify whether pain is consistent across time and affects the same subset of individuals, or changes over time either in its nature or with whom it affects. As such, the first objective of this study was to describe several pain characteristics in this population, including the frequency, intensity, duration, quality, and location of pain. Given the prevalence of pain in SB, it is also important to consider how youth with SB cope with pain as this can have implications for long-term adjustment. Research has consistently demonstrated associations between less adaptive forms of pain coping and adverse outcomes, such as increased pain intensity, functional disability, and poorer psychosocial functioning in pediatric pain populations (e.g., chronic migraine, chronic musculoskeletal pain, chronic abdominal pain; Claar, Baber, Simons, Logan, & Walker, 2008). However, to our knowledge, no study to date has examined how youth with SB typically cope with pain. Many studies have used the Pain Response Inventory (PRI; Walker, Smith, Garber, & Van Slyke, 1997), a well-established measure that assesses broad domains of coping (e.g., active, passive, accommodative). Therefore, the second objective of this study was to examine coping responses to pain among youth with SB, which were coded using the PRI framework. Finally, we examined longitudinal and bi-directional associations between pain and internalizing symptoms in youth with SB. In the larger pediatric literature, pain is strongly linked to reduced psychosocial functioning, particularly increased internalizing symptoms (Noel, Groenewald, Beals-Erickson, Gebert, & Palermo, 2016). Research examining the temporal relationship between chronic pain and internalizing symptoms has found evidence of bi-directional relations between the two constructs; some studies have found pain to precede the onset of internalizing symptoms (Noel et al., 2016), others have found it to follow the onset of internalizing symptoms (Shelby et al., 2013). Much of the literature on temporal relations between these constructs has also reported methodological limitations when making directionality claims (e.g., retrospective self-report). Therefore, this study sought to explore directionality with multi-informant longitudinal data. Determining the temporal relationship between these two constructs can inform clinical interventions in pediatric SB to prevent the development or maintenance of chronic pain and psychosocial difficulties. In line with trends in recent research, our hypothesis was that internalizing symptoms would predict later pain symptoms and that pain symptoms would predict later internalizing symptoms in youth with SB. Materials and Methods Participants Participants were recruited from a larger ongoing longitudinal study examining family and peer relationships, neuropsychological functioning, and psychological adjustment in youth with SB (e.g., Psihogios, Murray, Zebracki, Acevedo, & Holmbeck, 2017). This study represents an analysis of psychosocial functioning and pain symptoms during the first two time points, with each time point spaced 2 years apart. Families of youth with SB were recruited from four hospitals and a statewide SB association in the Midwest. Families were recruited in person at regularly scheduled clinic visits or through recruitment letters. Interested families were screened by phone or in person by a trained member of the research team to determine if their child met the following inclusion criteria: (a) a diagnosis of SB (types included myelomeningocele, lipomeningocele, and myelocystocele); (b) age 8–15 years; (c) proficiency in English or Spanish; (d) involvement of at least one primary caregiver; and (e) residence within 300 mi of the laboratory (to allow for data collection at participants’ homes). During recruitment a total of 246 families were approached, of which 163 agreed to participate. However, 21 of the 163 families could not be contacted or later declined to participate, and two families did not meet inclusion criteria. The final sample included 140 families of children with SB at Time 1 (53.6% female, Mage = 11.40; see Table I). Youth of families who declined to participate did not differ from participants with respect to type of SB (myelomeningocele or other), χ2 (1) = .0002, p > .05, shunt status, χ2 (1) = .003, p > .05, or occurrence of shunt infections, χ2 (1) = 1.08, p > .05. Table I. Youth Demographic and Spina Bifida (SB) Information at Time 1 . Total M (SD) or N (%) . Participants 140 (100%) Age 11.43 (2.46) Gender: female 75 (53.6%) Primary language: Spanish 28 (20.0%) Race  Caucasian 74 (52.9%)  African-American/Black 19 (13.6%)  Hispanic/Latino 39 (27.9%)  Asian 2 (1.4%)  Bi-racial 6 (4.3%) SB type  Myelomeningocele 122 (87.1%)  Non-myelomeningocele 17 (12.1%)  Unknown/not reported 1 (0.7%) Lesion level  Thoracic 23 (16.4%)  Lumbar 69 (49.3%)  Sacral 41 (29.3%)  Unknown/not reported 7 (5.0%) Shunt: present 109 (77.9%) Intelligence quotient) 85.75 (19.54) Family socioeconomic status 39.12 (16.09) T1 anxious/depression (P) 54.97 (6.08) T1 withdrawn/depression (P) 56.18 (6.56) T2 anxious/depression (P) 54.11 (5.16) T2 withdrawn/depression (P) 55.30 (5.48) T1 anxious/depression (T) 56.22 (6.89) T1 withdrawn/depression (T) 57.34 (7.89) T2 anxious/depression (T) 55.03 (6.31) T2 withdrawn/depression (T) 57.04 (8.54) T1 depressive symptoms (C) 1.31 (.23) T2 depressive symptoms (C) 1.27 (.19) . Total M (SD) or N (%) . Participants 140 (100%) Age 11.43 (2.46) Gender: female 75 (53.6%) Primary language: Spanish 28 (20.0%) Race  Caucasian 74 (52.9%)  African-American/Black 19 (13.6%)  Hispanic/Latino 39 (27.9%)  Asian 2 (1.4%)  Bi-racial 6 (4.3%) SB type  Myelomeningocele 122 (87.1%)  Non-myelomeningocele 17 (12.1%)  Unknown/not reported 1 (0.7%) Lesion level  Thoracic 23 (16.4%)  Lumbar 69 (49.3%)  Sacral 41 (29.3%)  Unknown/not reported 7 (5.0%) Shunt: present 109 (77.9%) Intelligence quotient) 85.75 (19.54) Family socioeconomic status 39.12 (16.09) T1 anxious/depression (P) 54.97 (6.08) T1 withdrawn/depression (P) 56.18 (6.56) T2 anxious/depression (P) 54.11 (5.16) T2 withdrawn/depression (P) 55.30 (5.48) T1 anxious/depression (T) 56.22 (6.89) T1 withdrawn/depression (T) 57.34 (7.89) T2 anxious/depression (T) 55.03 (6.31) T2 withdrawn/depression (T) 57.04 (8.54) T1 depressive symptoms (C) 1.31 (.23) T2 depressive symptoms (C) 1.27 (.19) Note. M = Mean; SD = standard deviation; T1 = Time 1, T2 = Time 2. Anxious/Depressive and Withdrawn/Depressive subscales are parent and teacher report on the Child Behavior Checklist; Child-reported depressive symptoms are youth report on the Child Depression Inventory. P = Parent, T = Teacher, C = Child. Open in new tab Table I. Youth Demographic and Spina Bifida (SB) Information at Time 1 . Total M (SD) or N (%) . Participants 140 (100%) Age 11.43 (2.46) Gender: female 75 (53.6%) Primary language: Spanish 28 (20.0%) Race  Caucasian 74 (52.9%)  African-American/Black 19 (13.6%)  Hispanic/Latino 39 (27.9%)  Asian 2 (1.4%)  Bi-racial 6 (4.3%) SB type  Myelomeningocele 122 (87.1%)  Non-myelomeningocele 17 (12.1%)  Unknown/not reported 1 (0.7%) Lesion level  Thoracic 23 (16.4%)  Lumbar 69 (49.3%)  Sacral 41 (29.3%)  Unknown/not reported 7 (5.0%) Shunt: present 109 (77.9%) Intelligence quotient) 85.75 (19.54) Family socioeconomic status 39.12 (16.09) T1 anxious/depression (P) 54.97 (6.08) T1 withdrawn/depression (P) 56.18 (6.56) T2 anxious/depression (P) 54.11 (5.16) T2 withdrawn/depression (P) 55.30 (5.48) T1 anxious/depression (T) 56.22 (6.89) T1 withdrawn/depression (T) 57.34 (7.89) T2 anxious/depression (T) 55.03 (6.31) T2 withdrawn/depression (T) 57.04 (8.54) T1 depressive symptoms (C) 1.31 (.23) T2 depressive symptoms (C) 1.27 (.19) . Total M (SD) or N (%) . Participants 140 (100%) Age 11.43 (2.46) Gender: female 75 (53.6%) Primary language: Spanish 28 (20.0%) Race  Caucasian 74 (52.9%)  African-American/Black 19 (13.6%)  Hispanic/Latino 39 (27.9%)  Asian 2 (1.4%)  Bi-racial 6 (4.3%) SB type  Myelomeningocele 122 (87.1%)  Non-myelomeningocele 17 (12.1%)  Unknown/not reported 1 (0.7%) Lesion level  Thoracic 23 (16.4%)  Lumbar 69 (49.3%)  Sacral 41 (29.3%)  Unknown/not reported 7 (5.0%) Shunt: present 109 (77.9%) Intelligence quotient) 85.75 (19.54) Family socioeconomic status 39.12 (16.09) T1 anxious/depression (P) 54.97 (6.08) T1 withdrawn/depression (P) 56.18 (6.56) T2 anxious/depression (P) 54.11 (5.16) T2 withdrawn/depression (P) 55.30 (5.48) T1 anxious/depression (T) 56.22 (6.89) T1 withdrawn/depression (T) 57.34 (7.89) T2 anxious/depression (T) 55.03 (6.31) T2 withdrawn/depression (T) 57.04 (8.54) T1 depressive symptoms (C) 1.31 (.23) T2 depressive symptoms (C) 1.27 (.19) Note. M = Mean; SD = standard deviation; T1 = Time 1, T2 = Time 2. Anxious/Depressive and Withdrawn/Depressive subscales are parent and teacher report on the Child Behavior Checklist; Child-reported depressive symptoms are youth report on the Child Depression Inventory. P = Parent, T = Teacher, C = Child. Open in new tab Data were collected at Time 2 for 111 (79%) of the original 140 participants. Reasons for attrition at Time 2 (N = 29) were as follows: 16 participants declined to participate, 12 participants were unable to be contacted, and 1 participant was deceased. Youth of families who did not participate at Time 2 did not differ from participants with respect to sex, χ2 (1) = .28, p > .05, socioeconomic status (SES), t(128) = −1.86, p > .05, type of SB (myelomeningocele or other), χ2 (1) = 1.19, p > .05, lesion level, χ2 (1) = .72, p>.05, or shunt status, χ2 (1) = 2.73, p > .05. Procedure This study was approved by university and hospital Institutional Review Boards. Trained undergraduate and graduate student research assistants collected data during home visits that lasted ∼3 hr. At Time 1, two separate 3-hr home visits were conducted. At Time 2 only one 3-hr home visit was conducted. At home visits with families who primarily spoke Spanish (N = 28), at least one research assistant who was fluent in Spanish was present. Prior to each home visit, informed consent from parents and assent from children were obtained. At Time 1, 26 of the 140 participants were estimated to have an intelligence quotient (IQ) below 70. These youth were assented and filled out the questionnaires when possible. Youth who were nonverbal did not fill out the questionnaires. Research assistants were trained to judge the validity of their responses based on the consistency of their answers. If their answers were not considered valid, they were not included in analyses. This resulted in a total of four participants at Time 1 being excluded from analyses. Parents also completed releases of information to obtain data from medical charts, health professionals, and teachers. After obtaining releases, questionnaires were sent to participant’s current teachers. During data collection, family members completed questionnaires independently. Questionnaires were offered in both English and Spanish. When questionnaires were only available in English, members of the research team, who were native Spanish speakers, translated and back-translated the questionnaires. The questionnaires were counterbalanced to avoid order effects. Research assistants read questionnaires aloud to participants when requested or when the youth appeared to be having reading difficulties. The current study used youth-, parent-, and teacher-reported questionnaire data. Families received $150 and small gifts (e.g., logo t-shirts, pens, water bottles) as compensation for participation at each time point. Measures Demographics At Time 1, parents reported on family and youth demographic information, including age, gender, race/ethnicity, income, education, and employment. The Hollingshead Four Factor Index of SES was computed using parent’s education and occupation, with higher scores indicating higher SES (Hollingshead, 1975). Illness Severity At Time 1, parents completed the Medical History Questionnaire (MHQ; Holmbeck et al., 2003), which included questions about youth’s disease-specific medical information including bowel and bladder functioning, ambulation method (i.e. ankle–foot orthoses [AFOs], knee–ankle–foot orthoses [KAFOs], hip–knee–ankle–foot orthoses [HKAFOs], wheelchair, or no assistance), medications, frequency of medical care, and surgery history. In addition to the MHQ, data were collected from medical charts to assess type of SB (i.e. lipomeningocele, meningocele, or myelomeningocele), shunt status, and lesion level (i.e. sacral, lumbar, or thoracic). These variables were used to compute an illness severity index based on membership in specific groups: shunt status (no = 1, yes = 2), myelomeningocele (no = 1, yes = 2), lesion level (sacral = 1, lumbar = 2, thoracic = 3), and ambulation status (no assistance/AFOs = 1, KAFOs/HKAFOs = 2, wheelchair = 3). Illness severity scores ranged from 4 to 10, with higher scores indicating higher levels of severity (Hommeyer, Holmbeck, Wills, & Coers, 1999). Youth IQ At Time 1, youth were administered the Vocabulary and Matrix Reasoning subtests of the Wechsler Abbreviated Scale of Intelligence (Wechsler, 1999). These subtests were used to estimate a Full Scale IQ score. The vocabulary subtest consists of 42 items, which measure an individual’s expressive vocabulary, verbal knowledge, and fund of information. The matrix reasoning subtests consists of 35 items which measure a child’s nonverbal fluid reasoning and general intellectual ability. These subtests have demonstrated high levels of internal consistency for youth 6–16 years old (α = .89 for Vocabulary, α = .92 for Matrix Reasoning; Wechsler, 1999). Internalizing Symptoms Internalizing symptoms were measured using youth, mother, father, and teacher report. At Times 1 and 2, youth reported on 27 depressive symptoms on the Child Depression Inventory (CDI), a well-validated measure of depression (Kovacs, 1992); higher scores indicated greater symptomatology. The CDI demonstrated acceptable levels of internal consistency and Times 1 and 2 (α =.82 and .78). Parents completed the Child Behavior Checklist (CBCL) and teachers completed the Teacher Report Form (Achenbach & Rescorla, 2001), well-validated broad-based measures of youth psychosocial functioning. Parents whose primary language was Spanish completed the Spanish version of the CBCL; this questionnaire has demonstrated strong validity and reliability in Spanish, and is commonly used in contemporary research (Rescorla et al., 2007). This study used mother, father, and teacher report on two internalizing subscales from the CBCL: anxious/depressed and withdrawn/depressed at Times 1 and 2. The anxious/depressed scale demonstrated acceptable reliability across reporters and at both time points: mother (T1: α = .76, T2: α = .70), father (T1: α = .68, T2 = .70), and teacher (T1: α = .65, T2: α = .74). Further, the withdrawn/depressed scale also demonstrated acceptable reliability across reporters and at both time points: mother (T1: α = .72, T2: α = .69), father (T1: α = .76, T2: α = .70), and teacher (T1: α = .84, T2: α = .87). Pain Characteristics We examined a variety of youth-reported pain characteristics including intensity, frequency, duration, location, and quality over the last 3 months. Pain intensity was measured via youth report on a Visual Analog Scale; youth marked a point along a 10-cm line ranging from “no pain” to “worst pain ever” (see Palermo, Zebracki, Newman, & Singer, 2004). Pain intensity was categorized according to severity (e.g., 0.0–0.9: none/minimal, 1.0–3.9: mild, 4.0–6.9: moderate, and 7.0–10.0: severe; Breivik et al., 2008). Pain frequency was measured on a 6-point Likert-type scale, ranging from “0—less than once a month” to “5—daily” (Palermo, Valenzuela, & Stork, 2004). Youth were considered to have chronic pain if they reported experiencing pain one or more times per week over the past 3 months (i.e. corresponding to a rating of a 2 on the 0–5 scale; King et al., 2011). Pain duration was measured on a 4-point Likert-type scale, ranging from “0—<1 hr” to “3—all day” (Palermo, Valenzuela, et al., 2004). Youth also marked pain location, or where they experienced pain on a validated body outline, which showed an anterior and posterior view of the body (Savedra, Tesler, Holzemer, Wilkie, & Ward, 1989); up to four responses were coded for each participant. Finally, pain quality was measured by youth checking off which descriptor(s) (e.g., sharp, aching, stinging, etc.) best captured the nature of their pain. Coping Behaviors in Response to Pain Youth were also asked to list “what helps you feel better” when experiencing pain due to SB. This question resulted in brief written responses such as “watch TV”, “drink cranberry juice”, or “take medicine.” Up to three of these responses were written per participant (though many people wrote fewer than three; range = 1–3). Initially, these responses were categorized into 152 very specific behavior categories (e.g., drink cranberry juice, watch TV). Next, we categorized these 152 specific behaviors based on the factors of the PRI (Walker et al., 1997), a frequently used measure for assessing child and adolescent coping behaviors in response to pain. Although the PRI has not yet been used to code free-written responses in this manner, our team sought to draw from an established, validated, and widely used measure to guide our coding of youths’ coping responses to pain. Two raters (a clinical psychology graduate student and a clinical psychology professor) independently coded each of the 152 behaviors into one of the first-order factors (e.g., social support, massage, distract/ignore) based on a comparison of the behavior to the specific items on the PRI and their respective factors. Inter-rater kappa values ranged from .87 to .99 for each first-order factor, indicating a very high level of agreement between raters. In several instances (N = 45) raters coded a behavior into more than one coping category (e.g., “mom rubbing tummy” was coded as both massage and social support). When there was disagreement, both reviewers discussed their response and came to a consensus. Although the coders categorized all of the responses from the entire sample, descriptive analyses are limited to the chronic pain subsample because the PRI was intended for use with pediatric chronic pain samples. Statistical Treatment Prior to hypothesis testing, psychometric properties were evaluated and composite scores were created when possible to reduce the number of analyses and decrease the possibility of shared method variance.1 Associations between measures in which there were two reporters (e.g., mother report, father report) were calculated using Pearson correlation coefficients; associations between three or more informants were calculated using Cronbach’s alpha coefficients (e.g., youth, mother, father, teacher). Criteria of r ≥ .40 and α > .60 were used, respectively, to determine which measures could be collapsed across reporters (Holmbeck, Li, Schurman, Friedman, & Coakley, 2002). Parent, youth, and teacher report of depressive symptoms (both anxious/depressed and withdrawn/depressed) were not highly associated. However, mother and father report of anxious/depressed and withdrawn/depressed symptoms were adequately correlated (r > .40) with each other at both time points and, therefore, were combined to create composite “parent report” internalizing scores at Times 1 and 2. Thus, parent, youth, and teacher report of internalizing symptoms were examined separately in the analyses. Descriptive statistics including mean, SD, and percentage frequency were used to address the first two aims of this study which involved describing the nature and prevalence of pain (Aim 1) and common behavioral coping responses (Aim 2). This study included descriptives at Times 1 and 2 for pain chronicity, intensity, and duration; all other descriptive statistics (location, quality, and coping responses) represent responses from Time 1 only. To address Aim 3, a series of hierarchical multiple linear and logistic regression analyses were conducted to examine associations between (a) internalizing symptoms at Time 1 and pain symptoms (intensity and chronicity) at Time 2, and (b) pain symptoms at Time 1 and internalizing symptoms at Time 2. The “pain chronicity” variable was created by dichotomizing the pain frequency variable into those who had chronic pain and those who did not have chronic pain (see above for criteria for determining those who have “chronic pain”). Logistic regressions were used when examining pain chronicity as an outcome. Several covariates were chosen due to their potential influence on increased risk of pain and internalizing symptoms, including older child age and female gender (Hyde, Mezulis, & Abramson, 2008; King et al., 2011), and lower SES (Dorner et al., 2011; Schreier & Chen, 2013). In addition, previous research has revealed associations between illness severity and psychosocial and physical functioning in this population (Hommeyer et al., 1999), thus we controlled for illness severity in analyses. Variables were entered in the following order: (Step 1) internalizing or pain symptoms at Time 1; (Step 2) covariates: illness severity, age, SES, gender, illness severity; and (Step 3) individual predictor (pain symptom [child report] or internalizing symptoms [teacher and parent report of anxious and withdrawn depressive symptoms, and child report of depressive symptoms]). Separate regressions were conducted for each combination of predictor and outcome variables. Missing data were handled with list-wise deletion. Results Description of Study Sample The total sample included 140 children and adolescents with SB ages 8–15, (53.6% female; 52.9% Caucasian), with the majority having myelomeningocele SB as well as a shunt. About half of the sample had a lumbar level lesion, the mean IQ was 85.68 (low average), and mean family SES was 39.44 (Table I) Associations between Time 1 pain symptoms and demographic variables revealed that lower IQ was associated with greater pain chronicity (r = −.22, p < .05). Further, gender was significantly associated with pain intensity at Time 1 (r = .27, p < .01), such that females with SB reported greater pain intensity than males with SB. No significant bivariate associations were found between age, SES, or race/ethnicity and Time 1 pain symptoms. With regard to illness-related variables, no significant bivariate associations were found between shunt status, lesion level, or illness severity and Time 1 pain symptoms. Objective 1: Describing the Nature of Pain in SB For the full sample at Time 1, 29.8% (N = 34) of youth-reported moderate or severe levels of pain. At Time 1, about 25% (N = 31) of the sample reported chronic pain (e.g., pain occurring at least once per week over the past 3 months), with over 10% of youth reporting daily pain (N = 13). Moreover, about 8% (N = 10) of the sample reported chronic pain at both Times 1 and 2 (see Table II). Pain frequency and intensity were similar at Time 2. Pain was predominately described as aching (42.4%, N = 50), and lasting less than an hour (55.8%, N = 43). The most common pain locations included back, extremities (e.g., hands, arms, legs), and abdomen, and head. See Table II for a description of pain characteristics for the full sample. Table II. Descriptive Data for Pain Frequency, Intensity, Duration, and Quality Variable . . Time 1 . Time 2 . . . N . % . N . % . Intensity  None/Minimal 45 39.5 44 43.1  Mild 35 30.7 33 32.3  Moderate 17 14.9 17 16.7  Severe 17 14.9 7 6.9 Frequency  No pain 34 27.3 32 30.0  Less than once per month 38 30.8 36 33.6  1–3 times per month 20 16.3 12 11.2  1 time per week 6 4.9 7 6.5  2–3 times per week 8 6.5 6 5.6  3–5 times per week 4 3.3 6 5.6  Daily 13 10.6 8 7.5  Chronic pain 31 24.8 27 25.2  Chronic pain at Times 1 and 2 10 8 Duration  <1 hr 43 55.8 38 52.1  A few hours 15 19.5 17 23.3  Half the day 13 16.9 10 13.7  All day 6 7.8 8 11.0 Pain quality  Aching 50 42.4  Dull 21 17.8  Sharp 19 16.1  Throbbing 17 14.4  Stinging 15 12.7  Pounding 13 11.0  Burning 10 8.5  Hammering 7 5.9  Cutting 6 5.1 Variable . . Time 1 . Time 2 . . . N . % . N . % . Intensity  None/Minimal 45 39.5 44 43.1  Mild 35 30.7 33 32.3  Moderate 17 14.9 17 16.7  Severe 17 14.9 7 6.9 Frequency  No pain 34 27.3 32 30.0  Less than once per month 38 30.8 36 33.6  1–3 times per month 20 16.3 12 11.2  1 time per week 6 4.9 7 6.5  2–3 times per week 8 6.5 6 5.6  3–5 times per week 4 3.3 6 5.6  Daily 13 10.6 8 7.5  Chronic pain 31 24.8 27 25.2  Chronic pain at Times 1 and 2 10 8 Duration  <1 hr 43 55.8 38 52.1  A few hours 15 19.5 17 23.3  Half the day 13 16.9 10 13.7  All day 6 7.8 8 11.0 Pain quality  Aching 50 42.4  Dull 21 17.8  Sharp 19 16.1  Throbbing 17 14.4  Stinging 15 12.7  Pounding 13 11.0  Burning 10 8.5  Hammering 7 5.9  Cutting 6 5.1 Note. M = Mean; SD = standard deviation; N = sample size. For pain quality, youth were able to check all descriptors that applied. Percentages reflect frequency of chosen descriptors. Pain intensity is presented according to severity categories (0.0–0.9: none/minimal, 1.0–3.9: mild, 4.0–6.9: moderate, and 7.0–10: severe). Open in new tab Table II. Descriptive Data for Pain Frequency, Intensity, Duration, and Quality Variable . . Time 1 . Time 2 . . . N . % . N . % . Intensity  None/Minimal 45 39.5 44 43.1  Mild 35 30.7 33 32.3  Moderate 17 14.9 17 16.7  Severe 17 14.9 7 6.9 Frequency  No pain 34 27.3 32 30.0  Less than once per month 38 30.8 36 33.6  1–3 times per month 20 16.3 12 11.2  1 time per week 6 4.9 7 6.5  2–3 times per week 8 6.5 6 5.6  3–5 times per week 4 3.3 6 5.6  Daily 13 10.6 8 7.5  Chronic pain 31 24.8 27 25.2  Chronic pain at Times 1 and 2 10 8 Duration  <1 hr 43 55.8 38 52.1  A few hours 15 19.5 17 23.3  Half the day 13 16.9 10 13.7  All day 6 7.8 8 11.0 Pain quality  Aching 50 42.4  Dull 21 17.8  Sharp 19 16.1  Throbbing 17 14.4  Stinging 15 12.7  Pounding 13 11.0  Burning 10 8.5  Hammering 7 5.9  Cutting 6 5.1 Variable . . Time 1 . Time 2 . . . N . % . N . % . Intensity  None/Minimal 45 39.5 44 43.1  Mild 35 30.7 33 32.3  Moderate 17 14.9 17 16.7  Severe 17 14.9 7 6.9 Frequency  No pain 34 27.3 32 30.0  Less than once per month 38 30.8 36 33.6  1–3 times per month 20 16.3 12 11.2  1 time per week 6 4.9 7 6.5  2–3 times per week 8 6.5 6 5.6  3–5 times per week 4 3.3 6 5.6  Daily 13 10.6 8 7.5  Chronic pain 31 24.8 27 25.2  Chronic pain at Times 1 and 2 10 8 Duration  <1 hr 43 55.8 38 52.1  A few hours 15 19.5 17 23.3  Half the day 13 16.9 10 13.7  All day 6 7.8 8 11.0 Pain quality  Aching 50 42.4  Dull 21 17.8  Sharp 19 16.1  Throbbing 17 14.4  Stinging 15 12.7  Pounding 13 11.0  Burning 10 8.5  Hammering 7 5.9  Cutting 6 5.1 Note. M = Mean; SD = standard deviation; N = sample size. For pain quality, youth were able to check all descriptors that applied. Percentages reflect frequency of chosen descriptors. Pain intensity is presented according to severity categories (0.0–0.9: none/minimal, 1.0–3.9: mild, 4.0–6.9: moderate, and 7.0–10: severe). Open in new tab For the chronic pain subsample, pain intensity was moderate to severe for 62.5% of the sample at Time 1. Moreover, a sizeable portion of the chronic pain subsample reported pain symptoms ranging in duration from a few hours to half of the day (40.8%, N = 11), with 14.8% of this subsample (N = 4) reporting experiencing pain all day. Pain was typically located in the extremities (30.8%, N = 20), back (26.2%, N = 17), and head (18.5%, N = 12), and was overwhelmingly characterized as aching (73.3%, N = 22). Objective 2: Youth Response to Pain It was possible to code responses into seven of the 13 original PRI’s primary subscales. These included condition-specific, rest, massage, problem solving, distraction, social support, and behavioral disengagement. For the chronic pain subscale, it was most common for youth to report using condition-specific methods when they experienced pain (57.7%; N = 15). Common condition-specific methods included over-the-counter pain medications (e.g., Tylenol and Advil) and drinking cranberry juice, which is frequently associated with preventing and treating urinary tract infections. Resting was the second most common method listed for ameliorating pain (30.8%, N = 8). Responses also revealed that youth with SB frequently use massage (23.1%, N = 6) and engage in problem solving (19.2%, N = 5) to address pain. Some youth also reported using distraction (15.4%, N = 4) or seeking social support (11.3%, N = 3). Finally, only one participant provided a response consistent with behavioral disengagement (3.8%). No participants generated methods that could be coded as self-isolation, stoicism, catastrophizing, acceptance, minimizing, or self-encouragement. Objective 3: Longitudinal, Bi-Directional Associations Between Pain and Psychosocial Functioning Hierarchical logistic and linear regressions revealed significant longitudinal associations between Time 1 internalizing symptoms and Time 2 pain symptoms. Assumptions were met for both logistic and linear regressions; logistic regressions passed the Hosmer and Lemshow Test and linear regressions did not demonstrate multicollinearity. Regression analyses included 5 predictors and 2 outcomes, resulting in a total of 10 analyses. After entering the covariates, greater teacher-reported anxious/depressed symptoms at Time 1 significantly predicted chronic pain group status at Time 2 [Exp(b) = 1.11, Wald = 5.14, p < .05; Table III], indicating that increased anxious/depressive symptoms increased the odds of being in the chronic pain group by a small but significant amount. Further, greater child-reported depressive symptoms at Time 1 significantly predicted higher pain intensity at Time 2 (β = .25, p < .05, ΔR2 = .05; Table IV), and accounted for about 5% of the variance, indicating again a small but significant effect. Although Time 1 child-reported depressive symptoms were significantly correlated with Time 2 chronic pain, in the logistic regression with covariates, this association was no longer significant (Table III). Of note, while few demographic and no illness-related variables were significantly associated with pain in bivariate correlations, significant associations were seen in longitudinal regressions. Specifically, age was related to chronic pain group membership, such that older youth at Time 1 were more likely to be in the chronic pain group at Time 2 (Table III). Further, illness severity was significantly associated with change in pain intensity at Time 2 in longitudinal regression, such that lower illness severity at Time 1 was significantly associated with greater pain intensity at Time 2 (Table IV). No significant associations were found between parent-reported depressive symptoms at Time 1 and chronic pain or pain intensity at Time 2 in longitudinal logistic and linear regressions, respectively (Table IV). With regard to examining the alternative direction, or associations between Time 1 pain symptoms and Time 2 internalizing symptoms, results revealed no significant associations. This finding was consistent across pain symptoms (chronic pain and pain intensity) and across reporters (parent, teacher, and child). Table III. Longitudinal Hierarchical Logistic Regression Analyses for Covariates and Teacher, Parent, and Child Depressive Symptoms at Time 1 Predicting Chronic Pain Group Membership at Time 2 Predictor . b . Exp (b) . Wald . Teacher report  Chronic pain (T1) .17 1.18 .04  Youth age .35 1.42 6.08*  Socioeconomic status (SES) .03 1.03 1.40  Illness severity −.30 .74 2.35  Gender .44 1.60 .42  Teacher-reported anxious/depressed symptoms (T1) .10 1.11 5.24*  Chronic Pain (T1) .62 1.86 .75  Youth Age .34 1.41 6.10*  SES .03 1.03 1.37  Illness Severity −.22 .80 1.46  Gender .51 1.67 .59  Teacher-reported withdrawn/depressed symptoms (T1) .03 1.03 .55 Parent report  Chronic pain (T1) .74 2.10 1.05  Youth age .38 1.45 7.26**  SES .02 1.02 .91  Illness severity −.24 .79 1.73  Gender .48 1.62 .57  Parent-reported anxious/depressed symptoms (T1) −.01 .99 .01 Chronic pain (T1) .74 2.10 1.10  Youth age .37 1.45 7.53**  SES .02 1.02 .93  Illness severity −.24 .79 1.74  Gender .49 1.63 .60  Parent-reported withdrawn/depressed symptoms (T1) −.01 .99 .03 Child report  Chronic pain (T1) .85 2.34 1.69  Youth age .40 1.49 8.28**  SES .03 1.03 1.66  Illness severity −.19 .83 1.25  Gender .48 1.61 .59  Child-reported depressive symptoms (T1) 1.81 6.12 1.17 Predictor . b . Exp (b) . Wald . Teacher report  Chronic pain (T1) .17 1.18 .04  Youth age .35 1.42 6.08*  Socioeconomic status (SES) .03 1.03 1.40  Illness severity −.30 .74 2.35  Gender .44 1.60 .42  Teacher-reported anxious/depressed symptoms (T1) .10 1.11 5.24*  Chronic Pain (T1) .62 1.86 .75  Youth Age .34 1.41 6.10*  SES .03 1.03 1.37  Illness Severity −.22 .80 1.46  Gender .51 1.67 .59  Teacher-reported withdrawn/depressed symptoms (T1) .03 1.03 .55 Parent report  Chronic pain (T1) .74 2.10 1.05  Youth age .38 1.45 7.26**  SES .02 1.02 .91  Illness severity −.24 .79 1.73  Gender .48 1.62 .57  Parent-reported anxious/depressed symptoms (T1) −.01 .99 .01 Chronic pain (T1) .74 2.10 1.10  Youth age .37 1.45 7.53**  SES .02 1.02 .93  Illness severity −.24 .79 1.74  Gender .49 1.63 .60  Parent-reported withdrawn/depressed symptoms (T1) −.01 .99 .03 Child report  Chronic pain (T1) .85 2.34 1.69  Youth age .40 1.49 8.28**  SES .03 1.03 1.66  Illness severity −.19 .83 1.25  Gender .48 1.61 .59  Child-reported depressive symptoms (T1) 1.81 6.12 1.17 Note. All predictor variables were measured at Time 1. The covariates of SES, youth age, illness severity, and gender were entered in as a block. Chronic pain at Time 1 was entered at Step 1. * p < .05, ** p < .01. Open in new tab Table III. Longitudinal Hierarchical Logistic Regression Analyses for Covariates and Teacher, Parent, and Child Depressive Symptoms at Time 1 Predicting Chronic Pain Group Membership at Time 2 Predictor . b . Exp (b) . Wald . Teacher report  Chronic pain (T1) .17 1.18 .04  Youth age .35 1.42 6.08*  Socioeconomic status (SES) .03 1.03 1.40  Illness severity −.30 .74 2.35  Gender .44 1.60 .42  Teacher-reported anxious/depressed symptoms (T1) .10 1.11 5.24*  Chronic Pain (T1) .62 1.86 .75  Youth Age .34 1.41 6.10*  SES .03 1.03 1.37  Illness Severity −.22 .80 1.46  Gender .51 1.67 .59  Teacher-reported withdrawn/depressed symptoms (T1) .03 1.03 .55 Parent report  Chronic pain (T1) .74 2.10 1.05  Youth age .38 1.45 7.26**  SES .02 1.02 .91  Illness severity −.24 .79 1.73  Gender .48 1.62 .57  Parent-reported anxious/depressed symptoms (T1) −.01 .99 .01 Chronic pain (T1) .74 2.10 1.10  Youth age .37 1.45 7.53**  SES .02 1.02 .93  Illness severity −.24 .79 1.74  Gender .49 1.63 .60  Parent-reported withdrawn/depressed symptoms (T1) −.01 .99 .03 Child report  Chronic pain (T1) .85 2.34 1.69  Youth age .40 1.49 8.28**  SES .03 1.03 1.66  Illness severity −.19 .83 1.25  Gender .48 1.61 .59  Child-reported depressive symptoms (T1) 1.81 6.12 1.17 Predictor . b . Exp (b) . Wald . Teacher report  Chronic pain (T1) .17 1.18 .04  Youth age .35 1.42 6.08*  Socioeconomic status (SES) .03 1.03 1.40  Illness severity −.30 .74 2.35  Gender .44 1.60 .42  Teacher-reported anxious/depressed symptoms (T1) .10 1.11 5.24*  Chronic Pain (T1) .62 1.86 .75  Youth Age .34 1.41 6.10*  SES .03 1.03 1.37  Illness Severity −.22 .80 1.46  Gender .51 1.67 .59  Teacher-reported withdrawn/depressed symptoms (T1) .03 1.03 .55 Parent report  Chronic pain (T1) .74 2.10 1.05  Youth age .38 1.45 7.26**  SES .02 1.02 .91  Illness severity −.24 .79 1.73  Gender .48 1.62 .57  Parent-reported anxious/depressed symptoms (T1) −.01 .99 .01 Chronic pain (T1) .74 2.10 1.10  Youth age .37 1.45 7.53**  SES .02 1.02 .93  Illness severity −.24 .79 1.74  Gender .49 1.63 .60  Parent-reported withdrawn/depressed symptoms (T1) −.01 .99 .03 Child report  Chronic pain (T1) .85 2.34 1.69  Youth age .40 1.49 8.28**  SES .03 1.03 1.66  Illness severity −.19 .83 1.25  Gender .48 1.61 .59  Child-reported depressive symptoms (T1) 1.81 6.12 1.17 Note. All predictor variables were measured at Time 1. The covariates of SES, youth age, illness severity, and gender were entered in as a block. Chronic pain at Time 1 was entered at Step 1. * p < .05, ** p < .01. Open in new tab Table IV. Longitudinal Hierarchical Multiple Linear Regression Analyses for Covariates and Teacher-, Parent, and Child-Reported Depressive Symptoms at Time 1 Predicting Pain Intensity at Time 2 Predictor . b . β . ΔR2 . Teacher report  Pain intensity (T1) .35 .31 .10*  Youth age .06 .14 .14  Socioeconomic status (SES) .01 .14 .14  Illness severity −.18 −.28 .14*  Gender .28 .14 .14  Teacher-reported anxious/depressed symptoms (T1) .01 .06 .00  Pain intensity (T1) .35 .31 .10**  Youth age .05 .14 .14  SES .01 .10 .14  Illness severity −.18 −.28 .14*  Gender .28 .14 .14  Teacher-reported withdrawn/depressed symptoms (T1) −.01 −.07 .00 Parent report  Pain intensity (T1) .35 .34 .11*  Youth age .06 .15 .13  SES .01 .14 .13  Illness severity −.16 −.26 .13*  Gender .45 .23 .13  Parent-reported anxious/depressed symptoms (T1) .03 .17 .02  Pain intensity (T1) .36 .34 .11**  Youth age .06 .15 .13  SES .01 .14 .13  Illness severity −.16 −.26 .13*  Gender .36 .19 .13  Parent-reported withdrawn/depressed symptoms (T1) .01 .05 .00 Child report  Pain intensity (T1) .35 .34 .12**  Youth Age .05 .11 .12  SES .01 .19 .12  Illness severity −.14 −.23 .12  Gender .36 .18 .12  Child-reported depressive symptoms (T1) 1.22 .23 .05* Predictor . b . β . ΔR2 . Teacher report  Pain intensity (T1) .35 .31 .10*  Youth age .06 .14 .14  Socioeconomic status (SES) .01 .14 .14  Illness severity −.18 −.28 .14*  Gender .28 .14 .14  Teacher-reported anxious/depressed symptoms (T1) .01 .06 .00  Pain intensity (T1) .35 .31 .10**  Youth age .05 .14 .14  SES .01 .10 .14  Illness severity −.18 −.28 .14*  Gender .28 .14 .14  Teacher-reported withdrawn/depressed symptoms (T1) −.01 −.07 .00 Parent report  Pain intensity (T1) .35 .34 .11*  Youth age .06 .15 .13  SES .01 .14 .13  Illness severity −.16 −.26 .13*  Gender .45 .23 .13  Parent-reported anxious/depressed symptoms (T1) .03 .17 .02  Pain intensity (T1) .36 .34 .11**  Youth age .06 .15 .13  SES .01 .14 .13  Illness severity −.16 −.26 .13*  Gender .36 .19 .13  Parent-reported withdrawn/depressed symptoms (T1) .01 .05 .00 Child report  Pain intensity (T1) .35 .34 .12**  Youth Age .05 .11 .12  SES .01 .19 .12  Illness severity −.14 −.23 .12  Gender .36 .18 .12  Child-reported depressive symptoms (T1) 1.22 .23 .05* Note. All predictor variables were measured at Time 1. The covariates of SES, youth age, illness severity, and gender were entered in as a block. Pain intensity at Time 1 was entered at Step 1. * p < .05, ** p < .01. Open in new tab Table IV. Longitudinal Hierarchical Multiple Linear Regression Analyses for Covariates and Teacher-, Parent, and Child-Reported Depressive Symptoms at Time 1 Predicting Pain Intensity at Time 2 Predictor . b . β . ΔR2 . Teacher report  Pain intensity (T1) .35 .31 .10*  Youth age .06 .14 .14  Socioeconomic status (SES) .01 .14 .14  Illness severity −.18 −.28 .14*  Gender .28 .14 .14  Teacher-reported anxious/depressed symptoms (T1) .01 .06 .00  Pain intensity (T1) .35 .31 .10**  Youth age .05 .14 .14  SES .01 .10 .14  Illness severity −.18 −.28 .14*  Gender .28 .14 .14  Teacher-reported withdrawn/depressed symptoms (T1) −.01 −.07 .00 Parent report  Pain intensity (T1) .35 .34 .11*  Youth age .06 .15 .13  SES .01 .14 .13  Illness severity −.16 −.26 .13*  Gender .45 .23 .13  Parent-reported anxious/depressed symptoms (T1) .03 .17 .02  Pain intensity (T1) .36 .34 .11**  Youth age .06 .15 .13  SES .01 .14 .13  Illness severity −.16 −.26 .13*  Gender .36 .19 .13  Parent-reported withdrawn/depressed symptoms (T1) .01 .05 .00 Child report  Pain intensity (T1) .35 .34 .12**  Youth Age .05 .11 .12  SES .01 .19 .12  Illness severity −.14 −.23 .12  Gender .36 .18 .12  Child-reported depressive symptoms (T1) 1.22 .23 .05* Predictor . b . β . ΔR2 . Teacher report  Pain intensity (T1) .35 .31 .10*  Youth age .06 .14 .14  Socioeconomic status (SES) .01 .14 .14  Illness severity −.18 −.28 .14*  Gender .28 .14 .14  Teacher-reported anxious/depressed symptoms (T1) .01 .06 .00  Pain intensity (T1) .35 .31 .10**  Youth age .05 .14 .14  SES .01 .10 .14  Illness severity −.18 −.28 .14*  Gender .28 .14 .14  Teacher-reported withdrawn/depressed symptoms (T1) −.01 −.07 .00 Parent report  Pain intensity (T1) .35 .34 .11*  Youth age .06 .15 .13  SES .01 .14 .13  Illness severity −.16 −.26 .13*  Gender .45 .23 .13  Parent-reported anxious/depressed symptoms (T1) .03 .17 .02  Pain intensity (T1) .36 .34 .11**  Youth age .06 .15 .13  SES .01 .14 .13  Illness severity −.16 −.26 .13*  Gender .36 .19 .13  Parent-reported withdrawn/depressed symptoms (T1) .01 .05 .00 Child report  Pain intensity (T1) .35 .34 .12**  Youth Age .05 .11 .12  SES .01 .19 .12  Illness severity −.14 −.23 .12  Gender .36 .18 .12  Child-reported depressive symptoms (T1) 1.22 .23 .05* Note. All predictor variables were measured at Time 1. The covariates of SES, youth age, illness severity, and gender were entered in as a block. Pain intensity at Time 1 was entered at Step 1. * p < .05, ** p < .01. Open in new tab Discussion To date, pain in youth with SB has been understudied and underreported. Thus, the purpose of this study was to describe the nature of pain in SB, examine how youth cope with their pain, and examine longitudinal, bi-directional associations between pain and internalizing symptoms. Results revealed that a small but significant minority of youth with SB experience chronic pain. Those that experience chronic pain typically employ condition-specific methods to alleviate their pain. Further, this study demonstrated that internalizing symptoms often precede chronic pain and pain intensity, highlighting the need for interventions targeting youth psychosocial functioning and regular assessment of youth pain symptoms. This study examined a variety of pain characteristics in this population. The prevalence of chronic pain (25%) in this sample was similar to what has been found in the larger pediatric pain literature (King et al., 2011). Moreover, pain intensity predominately fell in the minimal to mild range; however, a significant portion of the full sample and the majority of the chronic pain subsample reported experiencing moderate to severe pain. Location of pain resembled that found in previous studies, with youth reporting pain to be predominately located in the extremities, back, abdomen, and head (Clancy et al., 2007). Further, this study examined quality of pain, an underreported construct. Both the full sample and chronic pain subsample predominately described pain as aching. Pain literature has suggested that this information can be of use to better understand the etiology of pain as different sources of pain are often associated with a differential quality (Schilder, Magerl, Klein, & Treede, 2018). Indeed, aching pain may be related to muscle overuse or stress on joints related to ambulation methods. Given the limited understanding of pain in SB, pain quality may help clinicians better understand the etiology of pain and develop an appropriate treatment plan. Overall, these results suggest that youth with SB are vulnerable to the development of chronic pain. SB is a complex condition accompanied by a myriad of physical sequelae that require constant management. As such, pain may be under-identified and undertreated compared with other more pressing symptoms. Increased screening and education efforts including patient, family, and provider awareness of the risk for chronic pain in pediatric SB is needed. Moreover, given that 8% of the sample experienced continued, unresolved chronic pain 2 years later, there is likely a subgroup of youth with SB suffering from disabling, persistent chronic pain who require more intensive pain treatment. Clearly, there is a need to increase prevention and intervention efforts in this pediatric population. Regarding the relationship between demographic variables and pain symptoms, our findings highlight that females, older children/adolescents, and those with cognitive impairments may be at higher risk for experiencing pain symptoms. Notably, lower intellectual functioning was associated with pain chronicity. There are several possible reasons for this finding. It is possible that those with a lower IQ over-endorsed or misunderstood this question. On the other hand, youth with cognitive impairments have been found to experience pain more frequently than their TD peers (Breau, Camfield, McGrath, & Finley, 2003). Moreover, the current literature has also demonstrated associations between chronic pain and reduced attention and executive functioning (Moriarty, McGuire, & Finn, 2011), both of which are critical components of cognitive functioning and are often reduced in youth with SB (Rose & Holmbeck, 2007). Overall, these findings indicate that clinicians should be aware of these demographic risk factors for pain. Teaching health care professionals how to assess pain in populations with cognitive impairments such as SB may be particularly critical (e.g., with behavioral observation measures; Cohen et al., 2008). This was also the first study to examine how youth with SB cope with chronic pain symptoms. Results revealed that condition-specific methods were used most frequently, followed by resting, massage, problem solving, and distraction. Methods were primarily active, which is not surprising given that the prompt asked youth to describe what makes their pain feel better, rather than asking them how they cope with pain. Active strategies tend to be more behavioral where the person is doing something, whereas accommodative and passive strategies are frequently cognitive in nature (Shirkey, Smith, & Walker, 2011). However, the more frequent use of condition-specific methods may reflect a sort of pragmatism to coping with pain, which may be due to a perception of pain as only condition-specific in nature. Coupled with our findings indicating there were few significant relations between pain and condition-related characteristics and identifying internalizing symptoms as a predictor of pain intensity and frequency, these results suggest a greater need for children with SB and their families to be taught psychological-based strategies for pain management. Skills derived from cognitive-behavioral (e.g., deep breathing) and Acceptance and Commitment-based Therapy skills (e.g., mindfulness, acceptance) are effective methods for the treatment of chronic pain (Gauntlett-Gilbert, Connell, Clinch, & McCracken, 2013); however, it is possible that youth with SB and their families are not commonly taught these alternative pain management strategies due to the other rigorous demands of this medical condition. Indeed, biopsychosocial models of pain highlight the multifaceted, dynamic nature of pain (Liossi & Howard, 2016) and include condition-specific, intraindividual (psychological), and family (parenting) factors that exacerbate pain. Such factors have not been examined in depth in this population and deserve further attention in order to develop a comprehensive approach to treating pain in SB. Our longitudinal examination of pain symptoms and internalizing symptoms revealed that internalizing symptoms predicted the chronicity and future levels of intensity of pain in this sample, over and above demographic and clinical factors. These results indicate that an unexplored etiology of pain in SB may be the presence of depressive or anxious symptomology. Moreover, internalizing symptoms may serve to amplify otherwise acute and mild condition-related pain. Notably, associations were only found between teacher- and self-reported youth internalizing symptoms and pain. Similar findings involving teacher-reported data have been demonstrated in other research examining youth psychological functioning in SB (e.g., Stern, Driscoll, Ohanian & Holmbeck, 2018), possibly indicating that the connection between pain and psychosocial functioning is particularly salient in the school environment. In other words, health providers who rely heavily on parent report of youth psychosocial functioning may be missing important information about youth wellbeing and critical areas for early intervention. Therefore, it is imperative that stress, coping, and adjustment in the context of school be assessed by providers and parents, and that the interventions that focus on peer and school-based stress be developed for youth with SB. This study had several strengths, including the use of multiple methods and reporters of internalizing symptoms, comprehensive assessment of pain characteristics, and a longitudinal design. However, there were several limitations that should be noted. First, pain was only measured with youth report. Guidelines for best practice have recommended the use of multiple reporters, particularly for younger children and children with cognitive deficits (Cohen et al., 2008). Although research has consistently demonstrated strong associations between anxiety and pain (Shelby et al., 2013), this study unfortunately did not include specific measures of anxiety. Finally, while the longitudinal design is the strength of the study, our findings did not capture the day-to-day temporal relations among the variables of interest; future research may use daily diaries to advance understanding of potential bi-directional relationships between internalizing symptoms and pain in pediatric SB. Further, it is important to recognize that most regression analyses were not significant; therefore, findings should be interpreted with caution. Moreover, while having multiple reporters and measures was the strength of this study, findings were based on numerous regression analyses, increasing the likelihood of Type 1 error. Finally, effect sizes were small, indicating that internalizing symptoms are likely only one of several clinically significant factors to address in the prevention and treatment of pain in SB. There were also both strengths and limitations with our approach to measuring coping responses to pain. Although we coded written responses using the PRI as a theoretical framework (Walker et al., 1997), this measure is intended to be used in questionnaire format. Moreover, since the question that was asked focused on what one does that is helpful, most responses may have naturally fell into the active coping second-order factor. Relatedly, passive coping (e.g., catastrophizing), is typically linked to poorer outcomes, and thus we are likely missing important information about the use of passive strategies that youth with SB may understand to be unhelpful but use nonetheless. These concerns notwithstanding, this was still the first study to examine pain coping in this population. Therefore, this initial examination of pain coping in youth with SB may serve as a launching point for future studies to more systematically examine pain coping in youth with SB. Conclusions and Clinical Implications The results of this study have important clinical implications for promoting psychological and physical well-being in youth with SB. About 25% of youth with SB experience chronic pain and 10% experience daily pain. Pain is often understudied and undertreated in youth with SB, and there is a critical need for increased research and clinical attention to ameliorate pain in this population. Further, similar to the greater pediatric pain literature, internalizing symptoms are related to pain in this population, and may be key drivers of the development and chronicity of pain symptoms. Although pain in this population may be related to condition-specific factors, it also may be the result of poor psychological functioning, in line with biopsychosocial models of pain. Therefore, results from this study provide a critical first step in understanding the etiology of chronic pain in youth with SB, and underscore the importance of treatment for internalizing symptoms to ameliorate ongoing pain and suffering in children as they grow older. Future research should continue to expand our knowledge of the multifaceted etiology of pain in this population in order to facilitate the development of effective pain interventions for youth with SB and their families. Funding This research was supported in part by grants from the National Institute of Nursing Research and the Office of Behavioral and Social Sciences Research (R01 NR016235), National Institute of Child Health and Human Development (R01 HD048629), and the March of Dimes Birth Defects Foundation (12-FY13-271). This study is part of an ongoing, longitudinal study. Footnotes 1 Both parent and teacher report on the CBCL anxious/depression and withdrawn/depression scales, CDI total scores and pain intensity were mildly skewed at both time points. Analyses were run first with untransformed variables and then with square root transformations. Because findings were the same results presented here are with the untransformed variables. Acknowledgments The authors thank the Illinois SB Association as well as staff of the SB clinics at Ann & Robert H. Lurie Children’s Hospital of Chicago, Shriners Hospital for Children-Chicago, and Loyola University Medical Center. We also thank the numerous undergraduate and graduate research assistants who helped with data collection and data entry. Finally, we would like to thank the parents, children, and teachers who participated in this study. Correspondence regarding this manuscript can be sent to gholmbe@luc.edu. Conflicts of interest: None declared. References Achenbach T.M. , Rescorla L.A. ( 2001 ). 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This article is published and distributed under the terms of the Oxford University Press, Standard Journals Publication Model (https://academic.oup.com/journals/pages/open_access/funder_policies/chorus/standard_publication_model) Published by Oxford University Press on behalf of the Society of Pediatric Psychology 2020. This work is written by US Government employees and is in the public domain in the US.
TI - Longitudinal Associations Between Pain and Psychosocial Adjustment in Youth With Spina Bifida
JF - Journal of Pediatric Psychology
DO - 10.1093/jpepsy/jsaa037
DA - 2020-07-01
UR - https://www.deepdyve.com/lp/oxford-university-press/longitudinal-associations-between-pain-and-psychosocial-adjustment-in-toy0Cdsev0
SP - 673
EP - 684
VL - 45
IS - 6
DP - DeepDyve
ER -