Recurrent musculoskeletal pain in paediatric cerebral palsy : Relations to mental health, health-related quality of life and participation

Background Cerebral palsy (CP) is a disorder of movement and posture resulting from disturbances in the immature brain. Accompanying impairments including secondary musculoskeletal problems and mental health problems are common, and impairment is life-long. Thus, from a health care perspective, CP is an excellent model disease for asking what frames of reference should guide our understanding and evidence gathering about patient well-being, and what instruments should we use to assess these outcomes. Objectives The overall aim of the study was to explore how recurrent musculoskeletal pain relates to mental health problems, Health Related Quality of Life (HRQL) and participation in children with CP. Methods In study A, children with CP 8-18 years old were recruited from the South-West of Norway (n=75) and from the gait laboratory at Oslo University Hospital (n=78). All levels of motor impairment were represented. Clinical assessment, interview (child and parents together), and completion of questionnaires (child and parents separately) were performed. The interviewer asked for any kind of recurrent pain. If present, pain characteristics were explored and followed by professional judgement whether recurrent pain probably was of musculoskeletal origin and related to CP (RMP). Characteristics of RMP were regarded as localisation to muscles and/or joints, occurrence during or after exercise and dull or aching quality. Mental health problems, HRQL and participation were assessed by questionnaires (Strengths and Difficulties Questionnaire, Pediatric Quality of Life Inventory and Assessment of Life Habits respectively, in addition to General Health Questionnaire for parental mental health). In study B, children with CP 8-18 years (n=38) that started continuous intrathecal baclofen therapy (CITB) at Oslo University Hospital were assessed the day before implantation of a pump for baclofen delivery and after 6 and 18 months of treatment. Assessment consisted of clinical workup and parental interview. In cases of pain (33 children), this was assessed to be RMP before the child was accepted for CITB. Outcomes and explanatory variables throughout the study are discussed in relation to the ICF (International Classification of Function) that is WHO’s framework for measuring health and disability. Results In study A, 95 children (62 %) experienced RMP. Age above 14 years was the only significant predictor (OR 2.7). Children reported pain severity to be moderate. Parents reported pain to be more severe and with higher impact on sleep than their children did. Children and parents reported similar impact of pain on general activity and walking. Eighty-three pairs of children and mothers reported mental health problems and HRQL. Mothers in addition reported their own mental health. Self-reported mental health and HRQL were both better than proxy-reported. RMP was associated with more mental health problems and reduced HRQL in self-reports, but not in proxyreports. Mothers reported that more own mental problems were associated with more child mental problems and reduced child HRQL. Correlation (ICC) between mental health problems and psychosocial HRQL was 0.90 for both child and maternal report. One hundred and five parents reported child participation. RMP and more mental health problems were both associated with reduced participation. More parental mental health problems were associated with reduced parental satisfaction with the child’s accomplishment of daily activities. In study B, 35 children continued CITB for 18 months. Reduced pain and improved sleep occurred within 6 months of treatment. Social participation improved within 6 months and continued to improve until 18 months. Conclusions RMP is the main pain problem in paediatric CP. Systematic assessment of HRQL is suggested as a tool to broaden the scope of the consultation in (re)habilitation towards both pain and psychosocial issues. The child’s own perspectives on pain, mental health and HRQL should be recorded when possible in addition to that of the parents. The ICF concept of participation needs to be further clarified and operationalised; still the ICF reference frame is useful in both clinical work and research because of its comprehensiveness including the bio-psycho-social model and its non-categorical approach to health. Studies of children’s narratives on pain experiences and studies on self-reported mental health in CP are warranted

Hip pain in adolescents with cerebral palsy: a population-based longitudinal study

Aim To investigate the prevalence, characteristics, and risk factors of hip pain in adolescents with cerebral palsy (CP) and compare the findings with those of the same individuals 5 years earlier. Method Sixty-seven adolescents (28 females, 39 males; mean age 14y 7mo; SD 1y 5mo; range 12–17y) with bilateral CP, in Gross Motor Function Classification System (GMFCS) levels III to V enrolled in a CP surveillance programme were assessed for hip pain. Their caregivers responded to the questions on the intensity and frequency of hip pain from the Child Health Questionnaire (CHQ) (transformed to CHQ hip pain score; 100 indicates no pain). Interference of hip pain with daily activities and sleep was recorded on numeric rating scales. Hip displacement was measured radiographically by the migration percentage. Results Twenty-eight participants had 44 painful hips. Their mean CHQ hip pain score was 40 (SD 21.4; range 10–80). Independent risk factors for hip pain, low CHQ hip pain score, and interference with sleep were severe hip subluxation (migration percentage 50–89%) and GMFCS level V. A migration percentage of 50% to 89% was the only independent risk factor for interference with daily activities. Over 5 years, the number of participants with hip pain increased from 18 to 28, while the mean migration percentage of the most displaced hip was unchanged. Interpretation Our CP hip surveillance programme did not protect the participants against increasing prevalence of hip pain during adolescence. We suggest that surveillance programmes for CP should include guidelines on the characteristics and management of hip pain. What this paper adds Hip pain prevalence increased in adolescents over a 5-year period in a cerebral palsy surveillance programme. Risk factors for hip pain were Gross Motor Function Classification System level V and severe hip subluxation

Severe hip displacement reduces health-related quality of life in children with cerebral palsy: A population-based study of 67 children

Background and purpose — Hip displacement is common in children with severe cerebral palsy (CP) and can cause problems such as pain, contractures, and nursing difficulties. Caregiver priorities and child health index of life with disabilities (CPCHILD) is a recently developed measure of health-related quality of life (HRQL) in children with severe CP. The associations between CPCHILD scores and hip displacement have not been investigated. We explored the effect of hip displacement on HRQL. Patients and methods — 67 children were recruited from the population-based Norwegian CP register. Mean age was 9 (7–12) years. There were 40 boys. Gross motor function classification system (GMFCS) distribution was 12 level III, 17 level IV, and 38 level V. Hip displacement was assessed by radiographic migration percentage (MP). The criterion for hip displacement was MP of the worst hip of ≥40%. Primary caregivers responded to 5 of the 6 domains of the CPCHILD questionnaire. Results — Hip displacement was found in 18 children and it was significantly associated with lower scores on the CPCHILD domains 3 (Comfort and Emotions) and 5 (Health), but not with domains 1 (Activities of Daily Living/Personal Care), 2 (Positioning, Transfer, and Mobility), and 6 (Overall Quality of Life). GMFCS level V was a significant predictor of low scores in all the domains. Interpretation — For the assessment of HRQL in children with severe CP and hip problems, we propose a modified and simplified version of the CPCHILD consisting of 14 of 37 questions. This would reduce the responders’ burden and probably increase the response rate in clinical studies without losing important information

Recurrent pain in adolescents with cerebral palsy: a longitudinal population-based study

Aim To investigate the pain characteristics, pain interference with activities of daily living, and use of analgesics in adolescents with cerebral palsy (CP) and compare the results with previous findings. Method Sixty-seven adolescents (median age 14y 4mo, range 12y 2mo–17y, 28 females, 39 males) classified in Gross Motor Function Classification System (GMFCS) levels III to V, who participated in a CP surveillance programme, were assessed on pain measures twice, 5 years apart. Primary caregivers marked recurrent pain sites and graded pain interference with activities of daily living and sleep. Information on pain severity was obtained through two questions from the Child Health Questionnaire (CHQ) and were transformed into a pain score scaled from 0 to 100, where 100 represented no pain. The use of short-acting analgesics was recorded. Results Over 5 years, the prevalence of recurrent pain, number of pain sites, pain intensity, and pain frequency all increased significantly. The most frequent pain sites were the hip/thigh in GMFCS level V and knee in GMFCS level III. The median CHQ pain score decreased from 60 to 40 (p<0.001). Pain interference with activities of daily living increased (p=0.011) but not for sleep. Twenty-eight of 54 participants with moderate or severe pain (CHQ pain score ≤60) received no short-acting analgesics. Interpretation In adolescents with CP, pain increased over 5 years despite follow-up in a surveillance programme. For enhanced management of pain, we propose that an algorithm on pain should be included in surveillance programmes

The WE-Study: does botulinum toxin A make walking easier in children with cerebral palsy?: Study protocol for a randomized controlled trial

Background Intramuscular injections of botulinum toxin A (BoNT-A) have been a cornerstone in the treatment of spasticity for the last 20 years. In Norway, the treatment is now offered to two out of three children with spastic cerebral palsy (CP). However, despite its common use, the evidence for its functional effects is limited and inconclusive. The objective of this study is to determine whether BoNT-A makes walking easier in children with CP. We hypothesize that injections with BoNT-A in the calf muscles will reduce energy cost during walking, improve walking capacity, increase habitual physical activity, reduce pain and improve self-perceived performance and satisfaction. Methods/design This randomized, double-blinded, placebo-controlled, multicenter trial is conducted in a clinical setting involving three health regions in Norway. Ninety-six children with spastic CP, referred for single-level injections with BoNT-A in the calf muscles, will be invited to participate. Those who are enrolled will be randomized to receive either injections with BoNT-A (Botox®) or 0.9% saline in the calf muscles. Stratification according to age and study center will be made. The allocation ratio will be 1:1. Main inclusion criteria are (1) age 4 − 17.5 years, (2) Gross Motor Function Classification System levels I and II, (3) no BoNT-A injections in the lower limbs during the past 6 months and (4) no orthopedic surgery to the lower limbs during the past 2 years. The outcome measures will be made at baseline and 4, 12 (primary endpoint) and 24 weeks after injections. Primary outcome is change in energy cost during walking. Secondary outcomes are change in walking capacity, change in activity, perceived change in performance and satisfaction in mobility tasks, and pain. The primary analysis will use a linear mixed model to test for difference in change in the outcome measures between the groups. The study is approved by the Regional Ethical Committee and The Norwegian Medicines Agency. Recruitment started in September 2015. Discussion The evaluation of effect is comprehensive and includes objective standardized tests and measures on both impairment and activity level. Results are to be expected by spring 2019

Additional file 1: of The WE-Study: does botulinum toxin A make walking easier in children with cerebral palsy?: Study protocol for a randomized controlled trial

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Pain burden in children with cerebral palsy (CPPain) survey: Study protocol

Pain is a significant health concern for children living with cerebral palsy (CP). There are no population-level or large-scale multi-national datasets using common measures characterizing pain experience and interference (ie, pain burden) and management practices for children with CP. The aim of the CPPain survey is to generate a comprehensive understanding of pain burden and current management of pain to change clinical practice in CP. The CPPain survey is a comprehensive cross-sectional study. Researchers plan to recruit approximately 1400 children with CP (primary participants) across several countries over 6-12 months using multimodal recruitment strategies. Data will be collected from parents or guardians of children with CP (0-17 years) and from children with CP (8-17 years) who are able to self-report. Siblings (12-17 years) will be invited to participate as controls. The CPPain survey consists of previously validated and study-specific questionnaires addressing demographic and diagnostic information, pain experience, pain management, pain interference, pain coping, activity and participation in everyday life, nutritional status, mental health, health-related quality of life, and the effect of the COVID-19 pandemic on pain and access to pain care. The survey will be distributed primarily online. Data will be analyzed using appropriate statistical methods for comparing groups. Stratification will be used to investigate subgroups, and analyses will be adjusted for appropriate sociodemographic variables. The Norwegian Regional Committee for Medical and Health Research Ethics and the Research Ethics Board at the University of Minnesota in USA have approved the study. Ethics approval in Canada, Sweden, and Finland is pending. In addition to dissemination through peer-reviewed journals and conferences, findings will be communicated through the CPPain Web site (www.sthf.no/cppain), Web sites directed toward users or clinicians, social media, special interest groups, stakeholder engagement activities, articles in user organization journals, and presentations in public media