Examining tools for assessing the impact of chronic pain on emotional functioning in children and young people with cerebral palsy: stakeholder preference and recommendations for modification.

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.

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.

Chronic pain interference assessment tools for children and adults who are unable to self-report: A systematic review of psychometric properties.

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.

Upper limb impairments, process skills, and outcome in children with unilateral cerebral palsy.

AIM: To examine the relationships between upper limb impairments and independence in self-care (ISC) in children with unilateral cerebral palsy (CP). METHOD: One hundred and eight children with unilateral CP (46 females, 62 males; mean age 8y 7mo, SD 3y 9mo) recruited from a population register were assessed for upper limb muscle power, spasticity, sensation, motor control, and process skills, and for ISC as the functional outcome using structural equation modelling. RESULTS: The model showed good fit indices and explained 90% of the variance in ISC. Direct effects were significant between manual ability and ISC (ß=0.47), and process skills and ISC (ß=0.63). Sensation had a significant positive indirect effect on ISC through manual ability (ß=0.24) and a positive but marginally non-significant indirect effect through process skills (ß=0.21, bootstrapped 95% confidence interval -0.05 to 0.55). Spasticity had a significant negative indirect effect on ISC through its effect on manual ability (ß=-0.21). Age had a significant positive indirect effect on ISC, as did intellect, through their effect on process skills (ß=0.34 and 0.21 respectively). INTERPRETATION: ISC is affected by upper limb impairments and process skill. Sensation influences ISC through its effects on manual and process skill abilities. Both sensation and process skills require further evaluation to assist ISC in children with unilateral CP. WHAT THIS PAPER ADDS: Process skills and manual ability most strongly positively influence independence in self-care (ISC) in children with unilateral cerebral palsy. Sensation influences ISC through manual ability and process skill.

COMPROMISO DE EXTREMIDADES SUPERIORES, HABILIDADES DE PROCESAMIENTO Y PRONÓSTICO DE NIÑOS CON PARÁLISIS CEREBRAL UNILATERAL: OBJETIVO: Evaluar la relación entre el daño de las extremidades superiores y la independencia en el autocuidado de niños con parálisis cerebral unilateral. METODO: Ciento ocho niños con parálisis cerebral unilateral (46 mujeres, 62 varones; media de edad 8 años y 7 meses, desviación estándar 3 años y 9 meses) fueron reclutados de un registro poblacional. Se evaluó la fuerza muscular, espasticidad, sensibilidad, control motor y habilidades de procesamiento. Como resultado funcional para la independencia en el autocuidado se usaron modelos de ecuación estructural. RESULTADOS: El modelo mostro un adecuado ajuste y explicó el 90% de la varianza en la independencia en el autocuidado. Los efectos directos entre la habilidad manual y el autocuidado (ß=0,47), y las habilidades de procesamiento y el autocuidado (ß=0,63) fueron significativos. La sensibilidad tuvo un efecto positivo indirecto sobre el autocuidado a través de la habilidad manual (ß=0,24 y un efecto positivo, pero marginalmente no significativo a través de las habilidades de procesamiento (ß=0,21, error estándar 95% con un intervalo de confianza de -0,05 a 0,55). La espasticidad tuvo un efecto indirecto negativo significativo en el autocuidado, a través de su efecto en la habilidad manual (ß=−0,21). La edad tuvo un efecto positivo significativo indirecto sobre el autocuidado, al igual que en el intelecto, a través de su efecto sobre las habilidades de procesamiento (ß=0,34 y 0,21 respectivamente). INTERPRETACIÓN: La independencia en el autocuidado depende del compromiso de las extremidades superiores y de las habilidades de procesamiento. La sensibilidad influencia el autocuidado a través del efecto sobre la habilidad manual y las habilidades de procesamiento. Ambas, la sensibilidad y las habilidades de procesamiento requieren evaluación adicional para ayudar a la independencia de autocuidado en niños con parálisis cerebral unilateral.

DEFICIÊNCIAS NO MEMBRO SUPERIOR, HABILIDADES DE PROCESSAMENTO, E DESFECHO EM CRIANÇAS COM PARALISIA CEREBRAL UNILATERAL: OBJETIVO: Examinar as relações entre deficiências no membro superior e independência no auto-cuidado (IAC) em crianças com paralisia cerebral (PC). MÉTODO: Cento e oito crianças com PC unilateral (46 do sexo feminino, 62 do sexo masculino; média de idade 8a 7m, DP 3a 9m) recrutadas a partir de um registro populacional foram avaliadas quanto a força muscular, espasticidade, sensação, controle motor, e habilidades de processamento do membro superior, e quanto a IAC como resultado funcional, usando modelamento de equação estrutural. RESULTADOS: O modelo mostrou bons índices de adequação e explicou 90% da variância na IAC. Efeitos diretos foram significativos entre a capacidade manual e IAC (ß=0,47), e habilidades de processamento e IAC (ß=0,63). A sensação teve efeito significativo positivo indireto na IAC por meio da capacidade manual (ß=0,24) e efeito positivo indireto mas marginalmente não-significativo por meio das habilidades de processamento (ß=0,21, intervalo de confiança bootstrapped a 95% - 0,05 a 0,55). A espasticidade teve efeito significativo negativo indireto na IAC por meio do seu efeito na capacidade manual (ß=−0,21). A idade teve efeito positivo significativo indireto na AAC, assim como o intelecto, por meio do seu efeito nas capacidades de processamento (ß=0,34 e 0,21 respectivamente). INTERPRETAÇÃO: A IAC é afetada pelas deficiências do membro superior e pela habilidade de processamento. A sensação infuencia a IAC por meio de seus efeitos nas habiildades manuais e de processamento. Tanto a sensação quanto as habilidades de processamento requerem maior avaliação para facilitar a IAC em crianças com PC unilateral.

Clinician modifiable factors associated with better quality of life in children with acquired brain injury undergoing rehabilitation.

OBJECTIVES: To identify clinician-modifiable factors related to quality of life (QOL) in children with acquired brain injury (ABI). PARTICIPANTS AND METHODS: Thirty-nine children attending an ABI rehabilitation program (5-18 years) were assessed using the Personality Inventory for Children-2, Vineland Adaptive Behavior Scale-2, Handicap-Related Problems for Parents Inventory and Children's Assessment of Participation and Enjoyment. The Pediatric Quality of Life Inventory was completed by children and parents six months later. RESULTS: Children with lower levels of internalising and externalising behaviours, health and social skill problems, and higher family functioning had significantly higher levels of total QOL (child and parent rated) (r = -.47 to -.79). In addition, children with higher levels of adaptive behaviour had significantly higher parent rated total QOL (r = .46). Measures of mother's stressors had moderate but not statistically significant relationships with the child's total QOL (r = -.31 to -.35). There were moderate and statistically significant relationships between measures of participation in physical activities and total QOL as rated by children (r = .42-.48) but not parents (r = .11-.30). CONCLUSIONS: These findings suggest potential targets to be investigated in future clinical research in rehabilitation following ABI in children to optimise QOL.

A Single Session of Mirror-based Tactile and Motor Training Improves Tactile Dysfunction in Children with Unilateral Cerebral Palsy: A Replicated Randomized Controlled Case Series.

INTRODUCTION: This replicated randomized controlled crossover case series investigated the effect of mirror-based tactile and motor training on tactile registration and perception in children with unilateral cerebral palsy (UCP). METHODS: Six children with UCP (6-18 years; median 10 years, five male, three-left hemiplegia, four-manual ability classification system (MACS) I, one MACS II and one MACS III) participated. They attended two 90-minute sessions - one of mirror-based training and one of standard practice, bimanual therapy - in alternated order. Tactile registration (Semmes Weinstein Monofilaments) and perception (double simultaneous or single-point localization) were assessed before and after each session. Change was estimated using reliable change index (RCI). RESULTS: Tactile perception improved in four participants (RCI > 1.75), with mirror-based training, but was unchanged with bimanual therapy (RCI < 1.0 for all participants). Neither intervention affected tactile registration. DISCUSSION: Mirror-based training demonstrates potential to improve tactile perception in children with UCP. Copyright © 2016 John Wiley & Sons, Ltd.

CP or not CP? A review of diagnoses in a cerebral palsy register.

The purpose of this study was to document the inaccuracy rate of diagnosis of cerebral palsy recorded on the South Australian Cerebral Palsy Register. A total of 402 children born in South Australia from 1993 to 2002 and notified to the Register as having cerebral palsy were identified through the Register database, and 21 children (5.2%) were later identified to have a noncerebral palsy diagnosis. Of these, 5 had either a metabolic or a neurodegenerative disorder and 2 had a syndromic disorder (1 Joubert syndrome and 1 Sotos syndrome); the remaining 14 children had one of the following final diagnoses: developmental delay, gross motor delay, perinatal myositis, spinal subdural and subarachnoid arteriovenous malformation, and Erb's palsy. In 16 of 21 children (76%), the diagnosis was changed at 5 years of age or older. Studies based on population registers may need to take into account the possibility of misclassification, estimated to be at least 5.2% in this study. A complete clinical assessment at the time of diagnosis followed by regular reassessment would enable the clinician to exclude children with alternative diagnoses, which has important implications for clinical management and research based on cerebral palsy registers.

Self-esteem, self-concept, and quality of life in children with hemiplegic cerebral palsy.

OBJECTIVES: To investigate self-esteem, self-concept and quality of life in children with hemiplegic cerebral palsy (HCP) compared with typically developing peers. STUDY DESIGN: Cross-sectional evaluation of 86 children (3-16 years; 54 boys; mean age 9.4 +/- 3.7 years) with HCP and age and sex-matched peers. Self-esteem/concept was measured with the Self-Perception Profile for Children (age 8-16; n = 55 pairs) and the Pictorial Scale of Perceived Competence and Social Acceptance for Young Children (age 3-7 years; n = 31 pairs). Quality of life was measured with the Pediatric Quality of Life Inventory, version 4. RESULTS: Significant differences in mean scores ([95%CI] P < .05) favoring the peer group were found for physical competence (HCP 2.8 [2.5, 3.0]; peer 3.2 [3.1, 3.3]), athletic competence (HCP 2.7 [2.5, 2.9]; peer 3.1 [3.0, 3.3]), and scholastic competence (HCP 2.8 [2.6, 3.0]; peer 3.1 [3.0, 3.3]), but favored children with HCP for maternal acceptance (HCP 3.1 [2.9, 3.3]; peer 2.7 [2.5, 3.0]). Quality of life was significantly higher for the peer group for both parent (HCP 54.5 [51.1, 58.0]; peer 80.6 [78.3, 82.9]) and child (HCP 67.6 [62.7, 72.6]; peer 80.6 [78.1, 83.1]) scales. CONCLUSIONS: Children with HCP experience reduced quality of life and self-concept compared with typically developing peers.

Pain characteristics and their association with quality of life and self-concept in children with hemiplegic cerebral palsy identified from a population register.

OBJECTIVES: To describe and characterize the prevalence and quality of pain in a population-based community sample of children with hemiplegic cerebral palsy. METHODS: Outcomes were assessed from 2 domains of the World Health Organization International Classification of Functioning, Disability and Health: body structure/function (upper limb spans, modified Ashworth scale, Tardieu scale, sensory function), activities-participation (Assessment of Motor and Process Skills), Pediatric Quality of Life Inventory (PedsQL), and self-perception (Harter Self-Perception Profile). RESULTS: There were 107 participants of age (mean, 95% confidence interval) 8.94 years (8.23 to 9.65); 61 (57%) were boys and 58 (54%) had hemiplegia affecting the right side. Fifty-one (48%) reported pain. Pain mostly affected the involved side (n=28, 55%) and lower limb (n=19, 37%). Eighteen (35%) reported moderate to severe pain. Thirty-five (69%) said pain impacted on movement/activity and was of an aching quality (n=29, 57%). Mean (95% confidence interval) quality of life according to Pediatric Quality of Life Inventory was significantly lower for children experiencing pain compared with children experiencing no pain [parent 50.2 (45.9 to 54.5) vs. 60.1 (55.1 to 65.1), P<0.01; child 60.5 (55.4 to 65.6) vs. 75.8 (68.4 to 83.2), P<0.01]. The self-perception domains of Scholastic Competence [no pain, 3.02 (2.78 to 3.26); pain, 2.55 (2.31 to 2.79) P<0.01] and Behavioral Competence [no pain, 3.33 (3.07 to 3.60); pain, 2.88 (2.70 to 3.06) P<0.01] were significantly lower in children with pain aged > or =8 years. DISCUSSION: Pain is common in children with hemiplegic cerebral palsy with qualities suggesting a nociceptive origin. Pain is associated with lower quality of life and self-perception. Results suggest clinicians should assess and actively manage pain in this population.

Upper-limb botulinum toxin A injection and occupational therapy in children with hemiplegic cerebral palsy identified from a population register: a single-blind, randomized, controlled trial.

OBJECTIVES: The purpose of this work was to assess the effect of botulinum toxin A and occupational therapy compared with occupational therapy alone on body structure, activities participation, and self-perception in a sample of children (aged 3-16 years) with hemiplegic cerebral palsy recruited from a statewide register. PATIENTS AND METHODS: Participants of this single-blind, randomized, controlled trial identified from a population-based cerebral palsy register received either an individually prescribed and localized injection of botulinum toxin A with 4 sessions of occupational therapy over 4 weeks (intervention) or occupational therapy alone (control). Outcomes were assessed from 2 domains of the World Health Organization International Classification of Functioning, Disability, and Health: body structure (Modified Ashworth Scale and Tardieu Scale) and activities participation (Assessment of Motor and Process Skills, Goal Attainment Scale, Pediatric Evaluation of Disability Inventory, and Pediatric Quality of Life Inventory). Self-perception was also measured. RESULTS: All of the participants (intervention: n = 21; control: n = 22) provided data at baseline and 3 and 6 months. Mean age was 8.6 years; 23 were boys and 20 were girls. At 3 months, children allocated to receive the intervention performed significantly better in terms of body structure and activities participation. They reported improvements in self-perception for the global self-worth domain. At 6 months, the differences between the intervention and control groups persisted for the measures of body structure but not for activities participation or self-perception. CONCLUSION: Botulinum toxin A injection combined with a low-intensity occupational therapy program achieves significant improvements in body structure, activity participation, and self-perception.