Health-related quality of life in adolescents with cerebral palsy; a cross-sectional and longitudinal population-based study.

AIMS: The aims of this population-based cross-sectional and longitudinal study were to investigate different aspects of health-related quality of life (HRQoL) in adolescents with cerebral palsy (CP), to define possible changes in HRQoL from childhood to adolescence and to identify factors associated with low HRQoL in adolescence. METHODS: Proxy-reports of 64 adolescents, aged 12-17 years, with bilateral CP in GMFCS levels III-V participating in a surveillance programme, included five of the six domains from the HRQoL instrument Caregiver Priorities & Child Health Index of Life with Disabilities (CPCHILD): (1) Activities of Daily Living and Personal Care, (2) Positioning, Transfer and Mobility, (3) Comfort and Emotions, (5) General Health and (6) Overall Quality of Life, and the two questions on pain from the Child Health Questionnaire (CHQ). Fifty-eight participants comprised the longitudinal sample. RESULTS: From childhood to adolescence, the mean CPCHILD domain scores decreased slightly in General Health and remained unchanged in the other four domains. In the domain General Health, the number of medications increased, which was the reason for the score decrease. Pain severity increased significantly. Severe motor impairment was associated with low scores in domains 1, 2, 3 and 5, and more severe pain with low scores in domains 2, 3, 5 and 6. A low domain score in childhood was associated with a low score in each corresponding domain in adolescence. INTERPRETATION: An assessment of HRQoL should be included in CP surveillance programmes because this could identify needs for interventions in individuals with severe CP. This study indicates the importance of improved pain management in both children and adolescents with severe CP.

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.

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.

Daytime contacts and general practitioner consultations, and pain as a reason for encounter in children with cerebral palsy; a Norwegian national registry linkage study.

AIM: The aim of this study was to compare the prevalence of daytime contacts and consultations, and pain as a reason for encounter (RFE) with a general practitioner (GP), in children with cerebral palsy (CP) (cases) to that of the general paediatric population (controls). METHODS: The study linked the Norwegian Directorate of Health's database for the control and reimbursement of health expenses, and the Norwegian Quality and Surveillance Registry for Cerebral Palsy, including children born from 1996 to 2012 in the period 2006 to 2018. All daytime contacts were included. International Classification for Primary Care was applied for RFE. RESULTS: Cases accounted for 0.46% of all daytime contacts and 0.27% of all daytime consultations, the latter corresponding with the estimated national prevalence of CP. GPs registered more administrative contact and coded pain as an RFE less frequently in consultations with cases (6%) than with controls (12%). INTERPRETATION: Children with CP did not consult GPs more than the general paediatric population did. In consultations, GPs should ask for pain even if the child with CP or parent does not address pain. The local multidisciplinary team should encourage the family to consider consulting a GP if the child is in pain.KEY MESSAGESPrevalence of GP consultations in children with CP is similar to that of children in the general population.GPs perform more administrative work for children with CP than for their other paediatric patients.GPs code pain as an RFE less frequently in consultations with children with CP than in consultations with children in the general population.

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.

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. TRIAL REGISTRATION: ClinicalTrials.gov, NCT02546999 . Registered on 9 September 2015.

Severe hip displacement reduces health-related quality of life in children with cerebral palsy.

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.

Hip pain is more frequent in severe hip displacement: a population-based study of 77 children with cerebral palsy.

The aim of this study was to assess whether hip pain was associated with radiographic hip displacement (migration percentage, MP) in a population-based cohort of children with cerebral palsy. Seventy-seven children, mean age 9.5 (SD 1.6) years and Gross Motor Function Classification System level III-V, were assessed. Caregivers responded to the Child Health Questionnaire pain questions and located recurrent pain on a body map. Hip pain was reported in 22 children (29%) and 27 hips (18%). Hip pain was significantly more frequent in hips with MP more than or equal to 50%, in children with spastic quadriplegia, and in those with Gross Motor Function Classification System level V. We conclude that severe hip displacement with MP more than or equal to 50% was associated with hip pain, whereas slight or moderate subluxation did not influence the occurrence of such pain.

[Adolescents with cerebral palsy and their contact with the GP and the habilitative services]. / Ungdom med cerebral parese og deres kontakt med fastlege og habiliteringstjeneste.

BACKGROUND: Cerebral palsy (CP) often entails a need for health and social services throughout life. Upon scrutiny, it has been found that the transition from services for children and adolescents to services for adults is particularly challenging. The study investigates contact between adolescents with CP and their GPs/habilitative services up to and after the age of 18 years, and the percentage who have an individual habilitation plan (IHP). MATERIAL AND METHOD: An attempt was made to identify all those with CP born in 1992 and 1993 and resident in southeast Norway. Seventy-four patients were included (time point I, interview and clinical examination). Forty-two (57%) responded at the follow-up examination (time point II, questionnaire). RESULTS: At time point I, 35 (47%) of the adolescents had consulted their GP in the past year, 49 (66%) had been in contact with the habilitative services, and 42 (57%) had an IHP. Twenty-eight (38%) used mobility aids. Use of mobility aids gave lower odds of consultation with the GP. Longer distances from home to the habilitative services gave lower odds of contact with both the GP and the habilitative services. Five adolescents with mobility aids did not have an IHP. At time point II, the percentage who had consulted their GP had increased somewhat, the percentage who had been in contact with the habilitative services had reduced, and the percentage who had an IHP was unchanged. INTERPRETATION: The findings reinforce the assumption that the health services provided to disabled adolescents are less locally based and less well coordinated than is supposed, and that there may be geographical differences in the service offered.

Self-reported mental health in youth with cerebral palsy and associations to recurrent musculoskeletal pain.

PURPOSE: To explore self-reports on emotional, conduct, hyperactivity and peer problems and on prosocial behaviour in youth with cerebral palsy (CP), compare the reports to normative data and to mothers' reports and to analyse if recurrent CP-related musculoskeletal pain (RMP) influence the self-reports. METHODS: Eighty-one youth with CP (mean age 14.2 years, 40 boys) were assessed by clinical examination, interview and the questionnaire Strengths and Difficulties Questionnaire (SDQ) filled in by the youths and their mothers. Gross motor function (GMFCS) was: level I 43%, level II 41%, level III 12% and level IV-V 4%. RESULTS: Compared to normative data, youth with CP reported similar levels of peer problems, less conduct problems (p < 0.01), less hyperactivity problems (p < 0.01) and more prosocial behaviour (p < 0.01). Compared to the mothers' reports, youth with CP reported lower levels of peer problems (p < 0.01). Twenty-six boys (65%) and 28 girls (68%) had RMP. Girls, but not boys with RMP reported a higher level of peer problems (p = 0.02) than youth without RMP. CONCLUSION: Self-report on mental health in addition to parental proxy-report is important because it yields additional information. Peer acceptance and the impact of pain on peer problems and participation are candidate topics for further research.

The effect of interventions based on the programs of The Institutes for the Achievement of Human Potential and Family Hope Center.

OBJECTIVE: To compare the effect of the programs of IAHP and FHC with ordinary community-based programs. METHOD: Two-year observational study of two groups of children aged 2-15 years who were following the IAHP and FHC programs (N = 18) or community-based programs (N = 17), with additional material from interviews with parents, and a retrospective study (N = 9) based on file records and parent interviews. RESULTS: Changes in motor and cognitive function, language and behavior in the IAHP/FHC group well below the claims made by these programs, and few differences between this group and the comparison group. Intervention satisfaction lower prior to IAHP/FHC intervention than in the comparison group, and increased when moved to IAHP and FHC, independent of the children's progress. CONCLUSION: The substantial claims of superiority compared to other interventions made by IAHP and FHC are not supported, but parents appear to be met in a positive manner in these programs.

Parent-reported participation in children with cerebral palsy: the contribution of recurrent musculoskeletal pain and child mental health problems.

AIM: The aim of the study was to explore the contribution of recurrent musculoskeletal pain and mental health to aspects of participation in children with cerebral palsy (CP). METHOD: A total of 105 participants (54 males, 51 females); mean age 14 y (SD 3) were assessed by clinical examination, interview, and parental questionnaires. CP type distribution was as follows: spastic unilateral, 37%; spastic bilateral, 56%; and dyskinetic, 7%. Motor function assessed using the Gross Motor Function Classification System was level I, 33%; level II, 40%; level III, 15%; and level IV/V, 11%. Parents reported child participation using the Assessment of Life Habits, child mental health problems using the Strengths and Difficulties Questionnaire, their own mental health using the General Health Questionnaire, and their socioeconomic status. RESULTS: Recurrent musculoskeletal pain was associated with reduced accomplishment of daily activities (B=-1.22, p=0.02) and social roles (B=-1.17, p=0.03), and with reduced parental satisfaction with the accomplishment of daily activities (B=-1.14, p=0.03) and social roles (B=-1.48, p<0.01). Increasing levels of child mental health problems was associated with reduced accomplishment of daily activities (B=-0.10, p=0.02) and social roles (B=-0.11, p<0.01), and with reduced parental satisfaction with the accomplishment of social roles (B=-0.21, p<0.01). INTERPRETATION: Pain and child mental health should be considered in most participation models in CP. Regarding participation as an objective outcome measure is questionable.

Mental health, health related quality of life and recurrent musculoskeletal pain in children with cerebral palsy 8-18 years old.

PURPOSE: To extend knowledge on mental health with children's self-report and explore mental health and health related quality of life (HRQL) in a context of recurrent musculoskeletal pain in cerebral palsy (CP). METHOD: Eighty-three participants, mean age 14.2 ± 2.8 years, were assessed with clinical examination, interview and questionnaires. Gross motor function was GMFCS level I 42%, level II 42%, level III 12% and level IV-V 5%. Children self-reported mental health on SDQ (Strengths and Difficulty Questionnaire), HRQL on PedsQL (Pediatric Quality of Life), and pain on CHQ (Child Health Questionnaire). Mothers proxy-reported on the same questionnaires and reported own mental health on GHQ (General Health Questionnaire). RESULTS: Both self-reported mental health and HRQL was better than proxy-reported. Recurrent musculoskeletal pain was associated with more mental health problems and reduced HRQL in self-reports, but not in proxy-reports. CONCLUSIONS: In CP, the importance of child-report on mental health and HRQL when possible, in addition to parent proxy-report, cannot be overstated. Close co-operation between (re)habilitation and child psychiatry is urgent. Further research on self-reported mental health and impact of mental health problems is warranted together with a focus on the impact of recurrent musculoskeletal pain on participation.

Characteristics of recurrent musculoskeletal pain in children with cerebral palsy aged 8 to 18 years.

AIM: The aim of this study was to explore the prevalence, predictors, severity, and impact of recurrent musculoskeletal pain in children and adolescents with cerebral palsy (CP). METHOD: One hundred and fifty-three participants (81 males, 72 females) aged 8 to 18 years were assessed by clinical examination, interview, and questionnaires. CP type distribution was 38% unilateral spastic, 55% bilateral spastic, 6% dyskinetic, and 1% ataxic. Gross Motor Function Classification System (GMFCS) levels were as follows: level I, 54; level II, 56; level III, 20; level IV, 8; and level V, 15. Sixty-four children and 89 parents recorded pain on the Child Health Questionnaire, 56 children and 85 parents indicated impact of pain on 0 to 10 numeric rating scales, and 72 children indicated pain intensity on the Faces Pain Scale-Revised. RESULTS: Ninety-five (62%) children across all GMFCS levels experienced recurrent musculoskeletal pain. Age above 14 years was the only significant predictor (OR 2.90, 95% CI 1.22-7.80, p=0.02, adjusted for sex, CP type, gross motor function and mother's education). Children reported recurrent musculoskeletal pain 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. INTERPRETATION: Recurrent musculoskeletal pain is the dominating pain problem in children and adolescents with CP. Monitoring of musculoskeletal pain should be part of the medical follow-up across the whole range of motor impairment.

[Experiences with botulinum toxin injections against spasticity in children]. / Erfaringer med botulinumtoksininjeksjoner mot spastisitet hos barn.

BACKGROUND: Botulinum toxin injection is one of the newer options in the treatment of spasticity. Treatment with botulinum toxin is always combined with physiotherapy and often with casting. We show the extent to which botulinum toxin treatment has been taken into use in our department and discuss advantages and disadvantages of giving botulinum toxin injections at local hospitals. MATERIAL AND METHODS: 88 children with cerebral palsy aged 14 months to 16 years were treated with botulinum toxin between March 2000 and Dec. 2004. Injections were given in an outpatient setting, usually with the patient sedated with midazolam. Clinical examination after injection included assessment of spasticity and range of movement of joints. Motor function was videotaped. Side effects were continuously supervised. RESULTS: We performed 278 treatments; during the last year (2004) 7 treatments a month on average. 59 children were injected in lower limbs only, 14 were injected in upper limbs only, and 15 were injected in both upper and lower limbs. No serious side effects were recorded, neither from the botulinum toxin itself nor related to the injection procedure. INTERPRETATION: Assessment of indications for the use of botulinum toxin is now part of the medical follow up for children with spastic cerebral palsy. Treatment can safely be given at the paediatrics department in a local hospital. Injection in superficial muscles of the lower limbs is an easy task, while injection in small muscles and deep-seated muscles requires more special skills.

[Children with cerebral palsy referred to a multidisciplinary habilitation unit]. / Barn med cerebral parese henvist til en tverrfaglig habiliteringsenhet.

BACKGROUND: At Akershus University Hospital a new multidisciplinary team was set up to serve neurologically impaired children. We investigated how many children with cerebral palsy were referred to the team and whether referral resulted in an associated diagnosis. MATERIAL AND METHODS: All children with a diagnosis of cerebral palsy referred up until the end of the team's first year of full service were included. Data on birth weight and mobility were retrieved from patient files. Additional diagnoses were registered at inclusion date and two years later and compared to prevalence studies. RESULTS: 182 children had a diagnosis of cerebral palsy. 41 out of 114 children (36%) in the age bracket 6-14 were described as walking without using aids. After a minimum of two years' follow-up, 80 children had no neurological or psychiatric diagnosis (except cerebral palsy). INTERPRETATION: Children with mild motor impairment were underrepresented, and associated impairments, except epilepsy, were diagnosed less often than expected.

[Melatonin treatment of a blind child with serious sleep disorders]. / Melatoninbehandling av et blindt barn med store søvnforstyrrelser.

BACKGROUND: Children with developmental and neurological disabilities are prone to develop serious sleep-wake cycle disorders that may be difficult to treat. MATERIAL AND METHODS: Case history. RESULTS: A 5-year old blind boy with multiple disabilities developed a chronic sleep-wake cycle disorder as his main clinical problem. Treatment included introduction of strict sleep habits and strengthening of environmental "zeitgebers". After five months melatonin 3 mg was administered at night for 4 weeks. The observation period also included 3 weeks without melatonin. Sleep was registered prospectively by a sleep diary. Strict sleep habits combined with strengthening of "zeitgebers" partially improved the sleep problems, but did not establish a normal sleep pattern. When melatonin was added, he normalized his sleep pattern in a few days. His sleep problems returned during the weeks in which he did not receive melatonin. No side effects were observed. INTERPRETATION: Melatonin is a promising treatment alternative for serious sleep problems in blind children.

[Melatonin treatment of sleep disorders in disabled children]. / Melatoninbehandling ved søvnforstyrrelser hos funksjonshemmede barn.

BACKGROUND: There is a high prevalence of chronic sleep-wake cycle disorders in developmentally and neurologically disabled children. Such disorders are often resistant to hypnotic and psychotropic drugs. Administration of exogenous melatonin can have an effect upon the circadian rhythm and establish a normal sleep-wake cycle. MATERIAL AND METHODS: Medline, Premedline and EBM Reviews were searched for articles on melatonin treatment of sleep-wake cycle disorders in children. Additionally, sleep was registered systematically in 15 developmentally or neurologically disabled children treated with melatonin. RESULTS AND INTERPRETATION: The search identified 75 articles of which 33 included neurologically or developmentally disabled children. Reported results generally indicate that melatonin improves sleep-wake cycle disorders in these children. However, the majority of studies were open, and knowledge about side effects and long-term effects is limited. Our own experience is in accordance with the literature; melatonin had positive effects in 13 children. Suggestions for practical use of melatonin in disabled children are presented.