Aim
The primary aim of this review is to evaluate the evidence for pain prevalence in children and young adults with cerebral palsy. Secondary aims are to identify pain characteristics and types of ...pain measurement used in this population.
Method
Ovid MEDLINE, Embase, CINAHL Plus, and PubMed were searched in October 2016 and updated in November 2017. Two authors independently screened studies according to Preferred Items for Systematic Reviews and Meta‐Analyses (PRISMA) guidelines. Pain outcomes were categorized within a biopsychosocial pain framework, with pain prevalence extracted for all recall periods and measurement types.
Results
One hundred and six publications from 57 studies met inclusion criteria. Pain prevalence varied widely from 14 per cent to 76 per cent and was higher in females, older age groups, and those classified within Gross Motor Function Classification System level V. Pain was most frequent in the lower limbs, back, and abdomen and associated with reduced quality of life or health status. The influence of pain on psychological functioning, interference, and participation was inconclusive.
Interpretation
Variation exists in reported pain prevalence because of sampling bias, inconsistent measurement, varying recall periods, and use of different participant age ranges.
What this paper adds
Pain prevalence varies from 14 per cent to 76 per cent in children and young adults with cerebral palsy.
Pain is more prevalent in females, older age groups, and children in Gross Motor Function Classification System level V.
Resumen
Prevalencia y características del dolor en niños y adultos jóvenes con parálisis cerebral: una revisión sistemática
Objetivo
El objetivo principal de esta revisión es evaluar la evidencia de la prevalencia del dolor en niños y adultos jóvenes con parálisis cerebral. Los objetivos secundarios son identificar las características del dolor y los tipos de medición del dolor utilizados en esta población.
Método
Ovid MEDLINE, Embase, CINAHL Plus y PubMed se buscaron en octubre de 2016 y se actualizaron en noviembre de 2017. Dos autores seleccionaron de forma independiente los estudios de acuerdo con los criterios PRISMA. Los resultados del dolor se categorizaron dentro de un marco de dolor biopsicosocial, con una prevalencia de dolor extraída para todos los períodos de recuerdo y tipos de medición.
Resultados
Ciento seis publicaciones de 57 estudios cumplieron con los criterios de inclusión. La prevalencia del dolor varió ampliamente de 14% a 76% y fue mayor en mujeres, grupos de mayor edad y aquellos clasificados dentro del Sistema de Clasificación de la Función Motora Gruesa (GMFCS) level V. El dolor fue más frecuente en las extremidades inferiores, la espalda y el abdomen y se asoció con una reducción de la calidad de vida o estado de salud. La influencia del dolor en el funcionamiento psicológico, la interferencia y la participación no fue concluyente.
Interpretación
Existe una variación en la prevalencia del dolor reportado debido al sesgo de muestreo, la medición inconsistente, los diferentes períodos de recuerdo y el uso de diferentes rangos de edad de los participantes.
Prevalência e características da do rem crianças e jovens com paralisia cerebral: uma revisão sistemática
Objetivo
O objetivo primário desta revisão é avaliar a evidência para prevalência de dor em crianças e jovens com paralisia cerebral. Objetivos secundários incluem identificar características da dor e tipos de medidas para dor usadas nesta população.
Método
As bases Ovid MEDLINE, Embase, CINAHL Plus, e PubMed foram pesquisadas em outubro de 2016 e atualizada em novembro de 2017. Dois autores avaliaram os estudos independentemente de acordo com as diretrizes de Itens Preferidos para Revisões Sistemáticas e Metanálises (PRISMA). Os resultados de dor foram caracterizados em uma estrutura biopsicossocial, com a prevalência da dor extraída para todos os períodos e tipos de medida.
Resultados
Cento e seis publicações de 57 estudos atenderam aos critérios de inclusão. A prevalência da dor variou amplamente, de 14 a 76 por cento, sendo mais alta no sexo feminino, grupos mais velhos, e aqueles com classificação V no Sistema de Classificação da Função Motora Grossa (GMFCS). A dor foi mais frequente em membros inferiores, coluna, e abdomen, associada com redução na qualidade de vida ou estado de saúde. A influência da dor no funcionamento psicológico, interfêrencia, e participação foi inconclusiva.
Interpretação
Há variação nos rleatos de prevalência da dor por causa de vieses de recrutamento, medidas inconsistentse, períodos variados de busca, e uso de participantes em faixas etárias diferentes.
What this paper adds
Pain prevalence varies from 14 per cent to 76 per cent in children and young adults with cerebral palsy.
Pain is more prevalent in females, older age groups, and children in Gross Motor Function Classification System level V.
This article's has been translated into Spanish and Portuguese.
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Background Cerebral palsy (CP), the most common physical disability of childhood, is often accompanied by a range of comorbidities including pain. Pain is highly prevalent in children and young ...people with CP, yet has been poorly understood, inaccurately assessed, and inadequately managed in this vulnerable population. This narrative review presents recent research advances for understanding and managing pain in children and young people with CP, focusing on chronic pain, and highlights future research directions. Main body Pain prevalence rates in CP vary due to different methodologies of studies. Recent systematic reviews report up to 85% of children experience pain; higher in older children, females, and those with dyskinesia and greater motor impairment. Research examining the lived experience perspectives of children and their families demonstrate that even those with mild motor impairments have pain, children want to self-report pain where possible to feel heard and believed, and management approaches should be individualized. Notably, many children with cognitive and communication impairments can self-report their pain if adjustments are provided and they are given a chance. Past inadequacies of pain assessment in CP relate to a focus on pain intensity and frequency with little focus on pain interference and coping, a lack of tools appropriate for the CP population, and an assumption that many children with cognitive and/or communication limitations are unable to self-report. Recent systematic reviews have identified the most reliable and valid assessment tools for assessing chronic pain. Many were not developed for people with CP and, in their current form, are not appropriate for the spectrum of physical, communication, and cognitive limitations seen. Recently, consensus and co-design in partnership with people with lived experience and clinicians have identified tools appropriate for use in CP considering the biopsychosocial framework. Modifications to tools are underway to ensure feasibility and applicability for the spectrum of abilities seen. Conclusion Recent research advances have improved our understanding of the prevalence, characteristics and lived experience of chronic pain, and refined assessment methods in children and young people with CP. However, the very limited evidence for effective and novel management of chronic pain in this population is where research should now focus. Keywords: Cerebral palsy, Pain, Chronic pain, Biopsychosocial model
Purpose
To firstly identify tools for assessing the impact of chronic pain on emotional functioning in children and young people with cerebral palsy (CP), and secondly identify suggestions to improve ...their relevance, comprehensiveness, comprehensibility and feasibility for the CP population. Improving assessment of the impact of pain on emotional functioning can enhance quality of life by improving access to interventions for pain-related physical disability, anxiety and depression.
Methods
Ethics approval was granted through the Women’s and Children’s Health Network Human Research Ethics Committee (2022/HRE00154). A mixed methods study with people with lived experience and clinicians, and guided by the Consensus-based Standards for Measurement Instruments (COSMIN), was undertaken. An online survey identified the highest rated tools for validation and/or modification for young people with CP and chronic pain. Focus groups and interviews investigated content validity and feasibility of the tools identified as highest rated.
Results
The Fear of Pain Questionnaire for Children-SF (FOPQ-C-SF) and Modified Brief Pain Inventory (mBPI) were the highest rated for pain coping and multidimensional assessment (respectively) from the online survey (
n
= 61) of eight tools presented. Focus group and interview data (
n
= 30), including 58 unique modification suggestions, were coded to six categories: accessibility, comprehensibility, feasibility, relevance, presentation and comprehensiveness.
Conclusion
Potential modifications have been identified to improve the appropriateness and feasibility of the FOPQ-C-SF and mBPI for children and young people with CP. Future research should implement and test these modifications, prioritising the involvement of people with lived experience to ensure their needs are met alongside clinicians.
Plain english summary
Up to 75% of children and young people with cerebral palsy report chronic pain, which is much higher than those without cerebral palsy. Assessing how pain impacts emotional functioning, and how each individual copes with pain, is of particular importance due to known links between emotional functioning and long term pain outcomes. Reliable assessment of how pain impacts emotional functioning may also help to identify those who would benefit from psychological treatments. Although pain questionnaires are available, many are not suitable for children and young people with cerebral palsy with different communication, cognitive and movement abilities. This study had two aims: (1) to work out which of the currently available tools that assess how pain impacts emotional functioning are considered best for people with cerebral palsy, and (2) to identify potential modifications to these tools. The two most relevant and easy to understand questionnaires selected for modification were the Fear of Pain Questionnaire for Children and the modified Brief Pain Inventory. A number of modifications were identified, including improving how relevant the questions were to people with cerebral palsy, improving accessibility for people with complex communication needs or cognitive impairment and improving how easy to understand the questions and answer options are. These modifications can now be implemented to make it easier for people with cerebral palsy to use the pain assessments. They should then be tested in people with cerebral palsy with different communication, cognitive and movement abilities.
Aim
To systematically identify and evaluate the measurement properties of patient‐reported outcome measures (PROMs) and observer‐reported outcome measures (parent proxy report) of pain coping tools ...that have been used with children and young adults (aged 0–24 years) with a neurodevelopmental disability.
Method
A two‐stage search using MEDLINE, Embase, CINAHL, Web of Science, and PsycInfo was conducted. Search 1 in August 2021 identified pain coping tools used in neurodevelopmental disability and search 2 in September 2021 located additional studies evaluating the measurement properties of these tools. Methodological quality was assessed using the COnsensus‐based Standards for the Selection of Health Measurement INstruments (COSMIN) guidelines (PROSPERO protocol registration no. CRD42021273031).
Results
Sixteen studies identified seven pain coping tools, all PROMs and observer‐reported outcome measures (parent proxy report) versions. The measurement properties of the seven tools were appraised in 44 studies. No tool had high‐quality evidence for any measurement property or evidence for all nine measurement properties as outlined by COSMIN. Only one tool had content validity for individuals with neurodevelopmental disability: the Cerebral Palsy Quality of Life tool.
Interpretation
Pain coping assessment tools with self‐report and parent proxy versions are available; however, measurement invariance has not been tested in young adults with a neurodevelopmental disability. This is an area for future research.
Trihexyphenidyl for dystonia in cerebral palsy Harvey, Adrienne R; Baker, Louise B; Reddihough, Dinah Susan ...
Cochrane database of systematic reviews,
05/2018, Volume:
2018, Issue:
5
Journal Article
Peer reviewed
Open access
Background
Cerebral palsy occurs in up to 2.1 of every 1000 live births and encompasses a range of motor problems and movement disorders. One commonly occurring movement disorder amongst those with ...cerebral palsy is dystonia: sustained or intermittent involuntary muscle spasms and contractions that cause twisting, repetitive movements and abnormal postures. The involuntary contractions are often very painful and distressing and cause significant limitations to activity and participation.
Oral medications are often the first line of medical treatment for dystonia. Trihexyphenidyl is one such medication that clinicians often use to treat dystonia in people with cerebral palsy.
Objectives
To assess the effects of trihexyphenidyl in people with dystonic cerebral palsy, according to the World Health Organization's (WHO) International Classification of Functioning, Disability and Health (ICF) domains of impairment, activity and participation. We also assessed the type and incidence of adverse effects in people taking the drug.
Search methods
We searched CENTRAL, MEDLINE, Embase, eight other databases and two trials registers in May 2017, and we checked reference lists and citations to identify additional studies.
Selection criteria
We included randomised controlled trials comparing oral trihexyphenidyl versus placebo for dystonia in cerebral palsy. We included studies in children and adults of any age with dystonic cerebral palsy, either in isolation or with the associated movement disorders of spasticity, ataxia, chorea, athetosis and/or hypotonia. We included studies regardless of whether or not the study authors specified the method used to diagnose dystonia in their study population. Primary outcomes were change in dystonia and adverse effects. Secondary outcomes were: activity, including mobility and upper limb function; participation in activities of daily living; pain; and quality of life.
Data collection and analysis
We used standard methodological procedures expected by Cochrane.
Main results
We identified one study, which was set in Australia, that met the inclusion criteria. This was a randomised, double‐blind, placebo‐controlled, cross‐over trial in 16 children (10 boys and 6 girls) with predominant dystonic cerebral palsy and a mean age of 9 years (standard deviation 4.3 years, range 2 to 17 years). We considered the trial to be at low risk of selection, performance, detection, attrition, reporting and other sources of bias. We rated the GRADE quality of the evidence as low.
We found no difference in mean follow‐up scores for change in dystonia as measured by the Barry Albright Dystonia Scale (BADS), which assesses eight body regions for dystonia on a 5‐point scale (0 = none to 4 = severe), resulting in a total score of 0 to 32. The BADS score was 2.67 points higher (95% confidence interval (CI) −2.55 to 7.90; low‐quality evidence), that is, worse dystonia, in the treated group. Trihexyphenidyl may be associated with an increased risk of adverse effects (risk ratio 2.54, 95% CI 1.38 to 4.67; low‐quality evidence).
There was no difference in mean follow‐up scores for upper limb function as measured by the Quality of Upper Extremity Skills Test, which has four domains that collectively assess 36 items (each scored 1 or 2) and produces a total score of 0 to 100. The score in the treated group was 4.62 points lower (95% CI −10.98 to 20.22; low‐quality evidence), corresponding to worse function, than in the control group. We found low‐quality evidence for improved participation (as represented by higher scores) in the treated group in activities of daily living, as measured by three tools: 18.86 points higher (95% CI 5.68 to 32.03) for the Goal Attainment Scale (up to five functional goals scored on 5‐point scale (−2 = much less than expected to +2 = much more than expected)), 2.91 points higher (95% CI 1.01 to 4.82) for the satisfaction subscale of the Canadian Occupational Performance Measure (COPM; satisfaction with performance in up to five problem areas scored on a 10‐point scale (1 = not satisfied at all to 10 = extremely satisfied)), and 2.24 points higher (95% CI 0.64 to 3.84) for performance subscale of the COPM (performance in up to five problem areas scored on a 10‐point scale (1 = not able to do to; 10 = able to do extremely well)).
The study did not report on pain or quality of life.
Authors' conclusions
At present, there is insufficient evidence regarding the effectiveness of trihexyphenidyl for people with cerebral palsy for the outcomes of: change in dystonia, adverse effects, increased upper limb function and improved participation in activities of daily living. The study did not measure pain or quality of life. There is a need for larger randomised, controlled, multicentre trials that also examine the effect on pain and quality of life in order to determine the effectiveness of trihexyphenidyl for people with cerebral palsy.
Aim
To identify and evaluate psychometric properties of assessment tools for assessing pain interference in children, adolescents, and adults with chronic pain and the inability to self‐report.
...Method
The protocol was registered with PROSPERO (CRD42022310102). A search was run in MEDLINE, Embase, and PsycInfo (29th March 2022) to identify articles reporting psychometric properties of pain interference assessment tools for children, adolescents, and adults with chronic pain and the inability to objectively self‐report pain. Retrieved studies were reviewed by two authors (MGS, LCF) and study quality was assessed using COSMIN.
Results
Psychometric properties of 10 pain interference tools were assessed from 33 studies. The Paediatric Pain Profile (PPP) had low‐quality evidence for content validity and internal consistency with children and adolescents who are unable to self‐report. No tools for adults had evidence for content validity and internal consistency. No tool had evidence for all nine psychometric properties.
Interpretation
The PPP is recommended for pain interference assessment in children and adolescents with chronic pain and the inability to self‐report. Few tools are available for adults. Three tools for children (Patient‐Reported Outcome Measurement Information System Pediatric Proxy Pain Interference Scale; Bath Adolescent Pain Questionnaire for Parents; modified Brief Pain Inventory‐Proxy mBPI) and three tools for adults (Doloplus‐2; Patient‐Reported Outcome Measurement Information System Pain Interference Scale‐proxy; Brief Pain Inventory‐proxy) are promising but require further investigation.
This review systematically identified and evaluated the psychometric properties of pain interference assessment tools for children, adolescents, and adults with chronic pain and the inability to self‐report.
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PURPOSEChronic pain assessment tools exist for children, but may not be valid, reliable, and feasible for populations with functional, cognitive or communication limitations, for example, cerebral ...palsy (CP). This study aimed to (i) identify chronic pain assessment tools used with children and young people and rate their measurement properties; (ii) develop a CP specific feasibility rating tool to assess the feasibility of tools in CP; and (iii) categorise tools according to reporting method.MATERIALS AND METHODSAssessment tools were identified by literature review. Their measurement properties were rated using the COnsensus based standards for the Selection of health Measurement INstruments. The CP specific Feasibility Rating Tool was developed and used to rate the tools.RESULTSFifty-seven chronic pain assessment tools were identified. Six have content validity for CP, four of these use proxy-report. Forty-two tools were considered feasible for people with CP; 24 self report and 18 observational/proxy-report. Only the Paediatric Pain Profile has content validity and feasibility for people with CP unable to self-report.CONCLUSIONSThere are few valid, reliable and feasible tools to assess chronic pain in CP. Further research is required to modify tools to enable people with cognitive limitations and/or complex communication to self-report pain.
Aim
To evaluate pain prevalence and characteristics in children and adolescents with predominant dyskinetic and mixed (dyskinetic/spastic) cerebral palsy (CP) motor types.
Method
Seventy‐five ...participants with a diagnosis of CP and confirmed dyskinetic or mixed (dyskinetic/spastic) motor type took part in a multisite cross‐sectional study. The primary outcome was carer‐reported pain prevalence (preceding 2wks) measured using the Health Utilities Index‐3. Secondary outcomes were chronicity, intensity, body locations, quality of life, and activity impact.
Results
Mean participant age was 10 years 11 months (SD 4y 2mo, range 5–18y). There were 44 males and 31 females and 37 (49%) had predominant dyskinetic CP. Pain was prevalent in 85% and it was chronic in 77% of participants. Fifty‐two per cent experienced moderate‐to‐high carer‐reported pain intensity, which was significantly associated with predominant dyskinetic motor types (p=0.008). Pain occurred at multiple body locations (5 out of 21), with significantly increased numbers of locations at higher Gross Motor Function Classification System levels (p=0.02). Face, jaw, and temple pain was significantly associated with predominant dyskinetic motor types (p=0.005). Poorer carer proxy‐reported quality of life was detected in those with chronic pain compared to those without (p=0.03); however, chronic pain did not affect quality of life for self‐reporting participants.
Interpretation
Pain was highly prevalent in children and adolescents with predominant dyskinetic and mixed (dyskinetic/spastic) motor types, highlighting a population in need of lifespan pain management.
What this paper adds
Chronic pain prevalence in children and adolescents with predominant dyskinetic and mixed (dyskinetic/spastic) motor types is high.
Pain occurs across multiple body locations in predominant dyskinetic and mixed (dyskinetic/spastic) motor types.
Less recognized locations of pain include the face, jaw, and temple for predominant dyskinetic motor types.
What this paper adds
Chronic pain prevalence in children and adolescents with predominant dyskinetic and mixed (dyskinetic/spastic) motor types is high.
Pain occurs across multiple body locations in predominant dyskinetic and mixed (dyskinetic/spastic) motor types.
Less recognized locations of pain include the face, jaw, and temple for predominant dyskinetic motor types.
This article is commented on by Ferland on page 1233 of this issue.
Editor's Choice
Adequate management of pain is an ethical imperative. However, selected groups of individuals are particularly vulnerable to under‐recognition and under‐treatment of pain. Newborn infants are one of these important groups, as discussed in this month’s editorial.1 As documented by a number of studies, pain is a significant and indeed frequent problem in children with cerebral palsy (CP). My Editor’s Choice for the November issue highlights the particularly high prevalence of both acute and chronic pain in children with dyskinetic CP and those with mixed spastic and dyskinetic CP. It calls for systematic evaluation, effective treatment, and careful follow up to improve comfort throughout life in this group of children.
REFERENCES
1. Dan B. What do we know about pain in neonates? Dev Med Child Neurol 2020; 62: 1228.
Inconsistent and inadequate pain assessment practices in cerebral palsy (CP) have resulted from a lack of standardisation of pain assessment, limited use of appropriate tools and failure to integrate ...disability and biopsychosocial models. To assist with improving consistency, this study aimed to establish consensus from key stakeholders regarding domains considered essential for measuring chronic pain in children and young people with CP.
A modified electronic Delphi study was conducted on 83 stakeholders, including clinicians, researchers, people with CP and parents of children with CP. Participants rated 18 domains sourced from existing literature as either "core", "recommended", "exploratory" or "not required".
After two rounds of surveys, 12 domains were considered core: pain location, pain frequency, pain intensity, changeable factors, impact on emotional wellbeing, impact on participation, pain communication, influence on quality of life, physical impacts, sleep, pain duration and pain expression.
These domains reflect the complexity of pain in a heterogeneous population where medical comorbidities are common and communication and intellectual limitations impact significantly on the ability of many to self-report. The domains will be utilised to build a framework of pain assessment specific to children and young people with CP guided by the biopsychosocial model.
Implications for rehabilitation
Chronic pain is under-identified and poorly assessed in the cerebral palsy (CP) population.
The perspectives of clinicians, researchers and consumers are vital for developing a framework for chronic pain assessment in CP.
Consensus of key stakeholders found 12 domains considered essential to incorporate into a chronic pain assessment model in CP.
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