Site,frequency,and duration of pain in young children with spina bifida

2.1 Procedure and participants

This study was part of a multi-site study on development, health, and condition progression described elsewhere [30]. The data on pain were part of the larger study. Legal caregivers of children who were 3 years to 6 years and 11 months old, who have SB (International Statistical Classification of Diseases 9 codes of 741.0, SB with hydrocephalus and 741.9, SB without hydrocephalus), and were born between September 1, 2004, and August 31, 2009 were included, provided that caregivers were≥18 years old and English/Spanish-speaking. Eligible participants had to reside in one of the two catchment areas in Arizona and Utah. In Utah, children with SB were identified using population-based statewide surveillance data from the Utah Birth Defect Network. Parents of eligible children with SB who were attending the SB clinic during the study period were also invited to participate. Occasionally, eligible children with SB and their parents traveling from Idaho, Wyoming, and Nevada who were to attend the SB clinic in Salt Lake City were also invited to participate. In Arizona, eligible children with SB were identified through the birth defects monitoring program, hospital discharge databases, SB multispecialty clinics, and the primary children’s hospitals in Tucson and Phoenix to gather the eligible population, including health and demographic characteristics of the children. Three non-randomized options of participation were available: (1) in-person clinic visit that included neuropsychological testing of the child, caregiver surveys, and an administered survey (full protocol); (2) phone-administered survey (administered survey) and a mailed questionnaire (without neuropsychological testing; partial protocol 1); or (3) mailed questionnaire (without neuropsychological testing or administered caregiver survey; partial protocol 2). For Spanish-speaking participants, the consent, parental permission, and medical records release forms, parent interview, and parent surveys, which had been translated by translation services at Centers for Disease Control and Prevention, were conducted in Spanish. In Utah, a translator was available to assist for appointments with Spanish speakers. In Arizona, all appointments were conducted by bilingual professionals. In total, 152 individuals with SB were eligible, and 101 caregivers took part. Gift cards were given to caregivers who participated. Institutional review board (IRB) approvals were obtained at both sites.

Data were obtained from the administered study survey and medical records. Demographic information, level of lesion, type of SB, and comorbidities/conditions secondary to SB were extracted from the medical records.

2.2 Measures

3.2 Pain by sex, level of lesion, type of SB, and ambulation status (Aim 2)

3.3 Pain site, and number of painful body sites, by sex, level of lesion, type of SB, ambulation status, and comorbidity (Aim 3)

The head was the most commonly reported pain site, followed by the abdomen and the lower body. The number of pain sites ranged from zero to seven, with a median of two pain sites (Table 5). Painful body sites by sex, level of lesion, type of SB, ambulation status, and comorbidity are presented in Table 6 and the regions that were most reported are presented in Fig. 1. Pearson correlation coefficients indicated that the number of pain sites was positively correlated with more frequent complaints of pain, r(97) = 0.38, p < 0.001. As with the other pain outcomes, t-tests and ANOVAs did not support differences by sex (t(99) = –.22, p = 0.83), level of lesion (F(2, 95) = 2.66, p = 0.075), SB type (myelomeningocele versus non-myelomeningocele type, t(99) = –1.13, p = 0.260) or ambulation status (F(5,91)=0.77, p = 0.571) on number of pain sites.

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Fig. 1

Location of pain (percentage).

The pairwise correlations between how caregivers reported their own physical, mental, and emotional health and how they rated that of their children ranged from weak to moderate; that is, from r = 0.22 (child physical health and caregiver mental and emotional health) to r = 0.55 (caregiver physical health and caregiver mental and emotional health). All correlations were statistically significant (Table 7).

4.1 Overall frequency of pain

Seven out of ten children complained of pain, ranging from once a month or less to every day. Most (63%) reported that it had been experienced for up to three months, yet four out of ten reported that the pain had been experienced longer than three months. To study pain in more depth would require additional information beyond what was collected in this study. However, pain that lasts greater than three months, even if intermittent (most stated that the pain had lasted less than 24 hours) might be indicative of chronic pain.

4.2 Pain by sex, level of lesion, type of SB or ambulatory status

No statistically significant differences were found on complaints/duration of pain according to the child’s sex, level of lesion, type of SB, or ambulatory status. One reason for the null effects may be in the way that pain was measured in this study. The focus was on chronicity and frequency of pain. Potential differences in pain intensity or pain interference were not captured. For example, those who have MMC are likely to have hydrocephalus and might experience greater pain; however, this was not found in the analysis. As found here, individuals with MMC subtype may indeed experience pain with the same frequency as other subtypes, but the intensity of pain when it does occur could be greater for this group, though this hypothesis was not investigated in this study. The severity in which pain is experienced and its effect on functioning is an area for future research. In previous research on older children and adolescents with SB, greater pain was associated with reduced social activity involvement. Also, pain was indirectly associated with lower social competence [13]. Strong associations have been found between pain and lower health-related quality of life and higher depression scores [8]. That pain is not influenced by the predictors included in this study should be interpreted with caution, bearing in mind that it could be evaluated with greater depth and dimensionality [34] than what was captured in this study and by including a larger sample.

The results indicate that, according to the caregivers, pain is common across a population of young children with SB. These results are in line with previous research on pain in children with SB from different age groups. In a study of older children and adolescents with SB, 56%reportedly experienced pain once a week or more often [14]. Furthermore, level of lesion and ambulatory status were not significantly associated with pain [14]. Similar findings have been reported also in adults [27].

4.3 Body site

Headaches, pain in the abdomen, and pain in the lower body were reported most frequently. Headaches might be a sign of shunt dysfunction, and pain in the abdomen might be associated with constipation and urinary tract infections [35]. Clancy et al. [14] also reported the head and the abdomen as the most frequently reported painful body sites. Pain in the neck and shoulders were more commonly reported in older children [14] than in the current study. Surprisingly, children who had a shunt were not reported to have greater pain than those without a shunt. The null effect may be due to the lack of variation in the frequency of pain when it was reported. Fifty-three or 77%of the 69 who had pain indicated that their frequency of it was either once a month or less or more than once a month but less than once a week.

Recent findings on pain in adults with SB also support that pain in the upper body is more frequent [27]. In a Dutch study, young adults with SB and hydrocephalus reported significantly worse scores on body pain than a comparison group, with head, neck, and back being the most frequently reported sites [25]. Bellin and colleagues reported similar findings in terms of painful body sites [36] with shoulders, back, and head being most commonly reported in young adults with SB [36]. The variation in where the pain manifests –from pain in the lower body at younger ages to pain in the upper body in adulthood –might reflect a change in ambulation status over time. Young children might be more able or encouraged to walk, whereas later on in life they might be more likely to use a wheelchair. It might also represent differences in study designs or ambulatory status across different studies. Most of the children included in this study were ambulators/community ambulators, and most likely had sensation in their lower bodies. In the Oddson et al study [8], 38%of the children ambulated full-time. More than half of those who were reported experiencing pain had pain in more than one body site, and the number of pain sites was significantly positively correlated with more frequent complaints of pain.

4.4 Associations between physical and mental/emotional health

Caregivers who themselves reported worse scores on physical and mental health were more likely to report worse scores in their children. This is a cross-sectional study and cause and effect cannot be implied. To have a child with poor physical, mental, or emotional health might affect the health of the caregiver, or a caregiver who is not feeling well might be more likely to report that their child is also experiencing problems with their health.

4.5 Limitations

There were a number of limitations to the current study. MMC is a rare condition, which means that it is difficult to recruit large generalizable samples. This might result in studies that are under-powered. However, the study was population-based and attempts were made to recruit as many participants as possible. The measurement of pain here was frequency of pain complaints and duration. The degree to which complaints of pain is a good indicator of actual pain is unclear. The study also does not assess the intensity of pain or pain interference. More nuanced measures of pain that evaluate frequency, duration, and intensity would be more indicative of its adverse effect on the QoL. If and how pain interfered with participation was not included in the assessments. Severe chronic pain is quite different and likely would have other ramifications in terms of personal suffering and participation in school and leisure activities than would, for instance, mild, intermittent pain. Treatment modalities might also differ. Some of the items included were screening items or single items from validated measures. For instance, it would have been preferable to have used validated measures on caregivers’ self-rated mental and physical health. However, the scope of the study was already large and because it was not the main focus of the study this was not done.

An additional concern might be that proxies reported on the children’s pain. This decision was made not to overload the children who participated in the full protocol and because of the age of the target demographic. For those who participated in the partial protocols, it was not possible to reliably obtain child report on pain. The concordance between caregiver and child report of pain is a concern; both under-reporting and over-reporting are possible. In research on pediatric chronic pain, the concordance between maternal and child/adolescent agreement on pain location varied from fair to moderate agreement [37]. In the Clancy et al. study, the concordance between self- and parent report on child pain was the highest for location of pain [14]. Moreover, in younger children, the parents tended to underestimate the occurrence of the child’s current pain [14]. Also, it is possible that the correlations between child and parent “perceived health”, both reported by the caregiver, could be prone to a method effect, which could inflate the correlations. Almost all caregivers were mothers. It is possible that fathers would have reported on their children’s pain differently.

Although the intensity or the ramifications of the pain could not be determined, many of the young children included in the study seemed to deal with some pain. Playtime with peers is an important factor in children’s emotional and cognitive development. Experiences of pain are likely to reduce children’s desire to participate in developmentally appropriate activities. A physical disability, compounded with pain, can limit children’s participation in activities that enhance QoL, such as leisure activities and play. Cognitive challenges for some children with SB may make communicating pain experiences to their clinician or caregiver difficult and may require prompting from the clinician or caregiver. It should not be assumed that the individuals will mention the pain on their own accord. Difficulties with executive function or having gotten used to the pain might make it less likely for them to do so. Clinicians working with individuals with SB should be cognizant to inquire specifically about pain because pain in children with SB may be common, may be overlooked, and may reduce the QoL of those affected. Both self-report and proxy report of pain are strongly encouraged in clinical encounters.