Abstract
PURPOSE:
Caregivers provide unique insights into managing chronic pain in children and adolescents with dyskinetic and mixed dyskinetic/spastic cerebral palsy with communication limitations. This study explored the personal challenges caregivers face in supporting their child’s everyday pain management, including barriers and facilitators to effective chronic pain management.
METHODS:
Semi-structured interviews were undertaken with ten caregivers (all mothers) of children with either dyskinetic or mixed dyskinetic/spastic cerebral palsy. All children had chronic pain (> 3 months), were aged from 5 to 15 years, had significant functional limitations, and had either limited or no capacity to self-report their pain. Interpretative phenomenological analysis was used to explore caregivers’ subjective experiences of managing their child’s chronic pain within family, school, and healthcare contexts.
RESULTS:
Five superordinate themes emerged: 1. the continual challenge of problem solving pain and dyskinesia; 2. the pursuit of a solution; 3. unfulfilled preferences within pain management; 4. all-encompassing effects on families; and 5. the ongoing impacts of pain and dyskinesia with age.
CONCLUSION:
There is a need for structured pain education and resources targeted towards caregivers and support workers that account for the complex overlay of dyskinesia. There is a further need to ensure caregiver preferences for non-pharmacological pain treatments are met within family-centred care models.
Keywords
Introduction
Children with dyskinetic cerebral palsy (CP) account for a small proportion of the CP population (3%to 15%) and present with the complex overlay of dyskinesia [1]. Despite their small proportional representation, they form a complex subgroup of the CP population with high levels of functional motor impairment and frequent medical comorbidities including pain [2–4]. Pain is more common in children with CP classified within Gross Motor Functional Classification Scale (GMFCS) Level V and is caused by frequent comorbidities like scoliosis, hip displacement, muscle contracture, and gastro-intestinal disorders including constipation and gastro-oesophageal reflux disease [5–7]. Pain can also be caused by invasive interventional management (e.g., surgery), non-invasive interventional management (e.g., physiotherapy), general childhood conditions (e.g., headaches, period pain), and dyskinesia itself [8, 9]. Dyskinesia causes pain due to prolonged and repetitive muscle co-contractions and over time may lead to repetitive joint stress, poor body positioning in equipment, altered joint biomechanics, and possible early onset osteoarthritis [10]. Pain management in this population is further complicated by high existing care needs, use of multiple medications, the potential for life limiting conditions, and the presence of underlying communication difficulties [11–13]. Caregivers play a key role to ensuring pain is effectively managed in children with physical disability, and exploring their perspectives is pivotal to advancing pain management in this complex population [14, 15].
CP can be classified according to the predominant neuromotor type as either spastic, dyskinetic, or ataxic [1, 4]. However, there is increasing recognition that dyskinesia also commonly presents as a ‘mixed’ motor type in conjunction with spasticity, though ‘mixed’ is not a standalone CP classification [16]. Dyskinesia itself is reported to be under-recognized within the CP population, as historical methods of CP classification reportedly bias the classification of spastic CP motor types [17]. There is growing recognition that pain impacts children with both predominant dyskinetic and mixed dyskinetic/spastic CP motor types, with a need to better understand their unique pain experiences [18, 19].
Pain itself is recognized to be a complex experience influenced by a range of cognitive, affective, and behavioural mechanisms within bio-psychosocial pain models [20, 21]. Caregivers are likely to have a strong influence over their child’s own experiences of pain and disability and, in children with communication limitations, play a key role in interpreting their pain experiences [22, 23]. Caregivers are reported to have a high awareness of their child’s pain behaviours and physiological responses and are crucial to ensuring pain is identified and adequately treated in their child [14, 15]. For a subset of children with CP and communication limitations, the inability to self-report pain is constant throughout childhood. However, as CP is a heterogenous condition, some children with cognitive capacity may develop the ability to self-report pain with age, education, therapeutic interventions, and access to technology. For children and adolescents with CP and dyskinesia, specific barriers to effective pain management occur due to their communication limitations and high levels of physical impairment, with caregivers in a unique position to illuminate both their own needs and their children’s needs within pain management.
Despite research recognizing high chronic pain prevalence in children with dyskinetic CP, use of evidence based chronic pain management strategies within bio-psychosocial models is reported to be lacking in this population [18, 19, 24–26]. For example, in Australia psychological therapies are reported to be used infrequently to manage pain in children with CP, despite their strong evidence base in other populations with chronic pain [19, 27]. Caregivers play a key role in ensuring timely and effective pain management for chronic pain in their children. The purpose of this study is to explore the personal perspectives of caregivers regarding challenges faced within everyday pain management of their children with either dyskinetic or mixed dyskinetic/spastic CP, to ensure both caregiver and children’s needs are accounted for in pain management.
Methods
In-depth semi-structured interviews were undertaken with caregivers of children and adolescents with predominant dyskinetic and mixed spastic/dyskinetic CP who had limited capacity or were unable to self-report their pain. Interpretative Phenomenological Analysis (IPA) was employed to explore complex and emotionally laden experiences of chronic pain and disability to their fullest depth [28]. The study was part of a larger mixed methods study undertaken at The Royal Children’s Hospital and Monash Children’s Hospital, Victoria, Australia. Data collection occurred between January 2018 and March 2019 and was approved by the Royal Children’s Hospital, Monash University, and Monash Children’s Hospital Ethics Committees (HREC/17/RCHM/359).
Study procedures
Caregivers were identified from a larger multi-site, prospective, cross-sectional study (
Participants
As part of consenting procedures for the larger cross-sectional study, caregivers agreed to be approached to take part in this qualitative study if deemed eligible. A reasonably homogeneous sample of caregivers were purposively selected based on a range of outcomes and demographics collected in the quantitative study [18]. Parents were eligible if their children: 1) were classified within GMFCS level V; 2) had chronic pain (>3 months); 3) were unable or had limited capacity to self-report pain; and 4) had pain impacting their quality of life. Chronic pain was measured using the International Association for the Study of Pain definition of constant or recurrent pain over the previous 3 months [32]. Children’s quality of life was measured using the primary caregiver Cerebral Palsy Quality of Life Questionnaire child and teen versions [33]. Functional motor impairment was classified using GMFCS and communication effectiveness using the Communication Functional Classification System [34, 35]. Participants were the first 10 from the larger cross-sectional study who agreed to take part, an appropriate sample size for IPA, which recognizes the importance of the analytical accounts of individual cases.
Data generation
Qualitative data
Semi-structured interviews were conducted with caregivers by a single interviewer (CM) in a mutually convenient location, typically their own home. Interviews ranged in length from 45 to 90 minutes, were audio recorded with permission, and were transcribed verbatim. A semi-structured interview guide consisting of open-ended questions with prompts was developed by the research team with the research purpose in mind (Table 1). The interview guide was piloted with a mother of a child with dyskinetic CP and chronic pain. It was then altered in response to feedback regarding the nature and relevance of its contents. The interview guide was flexible in its structure, giving caregivers the opportunity to focus their interviews on pain conditions and concerns of their choice while the interviewer asked probing questions as emotive and important topics arose. Each interview commenced with caregivers discussing their child’s dyskinesia presentation and choosing appropriate terminology to reference when referring to dyskinetic movement disorder. Further questioning explored their child’s general medical care needs at school, home, and hospital to help give the interviewer context of the caregiver’s role. Subsequent questioning explored the daily impact of pain and dyskinesia on both the caregiver’s and child’s lives.
Sample interview questions
Please note use of the word ‘uncontrolled movements’ varied depending on caregiver preference for dyskinesia terminology. Health professionals referred to the child’s main treating medical professional and allied health workers. Non-medical referred to support staff either at school or home.
Sample interview questions
Please note use of the word ‘uncontrolled movements’ varied depending on caregiver preference for dyskinesia terminology. Health professionals referred to the child’s main treating medical professional and allied health workers. Non-medical referred to support staff either at school or home.
Two authors (CM, JW), one a physiotherapist (CM) and the other an occupational therapist (JW), independently coded data using interpretative phenomenology analysis [28]. This approach is ideographic in that each case is analysed in full before moving on to the next. Both authors commenced by reading transcripts multiple times to fully immerse themselves in the data and making initial notes and comments regarding content manually within the transcript margins. Initial notes were then developed into emergent thematic categories with all subsequent coding undertaken using NVivo 9. Authors compared and iteratively revised emergent thematic categories as they arose, with any differences in interpretations between authors resolved by negotiation and consensus. Thematic categories were supported by exemplar quotes and transcripts were re-read and cross-checked for the emergent categories. After each case had been analysed in full, processes of abstraction were used to search for patterns across all cases. Emergent themes were grouped into superordinate categories using a master table, which was then transformed into a narrative account.
Data trustworthiness was ensured by using the strategies of data immersion, reflexive analysis, field notes, and peer debriefing to ensure researchers remained open and unbiased during data analysis [36, 37]. The interviewer (CM) knew all caregivers from undertaking data collection within the larger cross-sectional study. The second coder (JW) was an experienced qualitative researcher with no background in CP management or knowledge of caregivers and their children. The use of two coders with different experience and knowledge of CP management meant that preconceived biases were easily identified using processes of open discussion and peer debriefing between coders. To ensure interpretations were well grounded within the data, member checking was completed by sending a summary of themes to each caregiver for their verification and confirmation of the interpretation. Only two caregivers responded, one via email and the other in person, reporting they were satisfied with the thematic interpretation, suggesting that no changes needed to be made.
Results
The baseline characteristics of the 10 caregivers and their children are presented in Table 2. To provide further context, children’s pain presentations and pharmacological and nonpharmacological treatments are presented in Table 3. Five key superordinate themes arose from subthemes within the data, and these are described below. Barriers and facilitators to pain management are further described in Table 4.
Baseline characteristics for caregivers and child
Baseline characteristics for caregivers and child
Baseline characteristics of the child’s pain presentation and pharmacological and nonpharmacological management
Health Utilities Index-3, 1 = No pain, 2 = Mild to moderate pain that prevents no activities, 3 = Moderate pain that prevents a few activities, 4 = Moderate to severe pain that prevents most activities, 5 = Severe pain that prevents all activities. Please note bodily sites of pain were based on the Childhood Arthritis and Rheumatology Research Alliance Body Diagram.
Caregivers’ perspectives of the barriers and facilitators to effective pain management in dyskinetic CP
This theme represents the ongoing challenges faced by caregivers in negotiating complex pain and dyskinesia paradigms and the strategies they use to accommodate these challenges within their everyday lives.
Caregivers perceived difficulty in identifying their child’s pain due to the numerous possible pain sources alongside the unpredictable overlay of dyskinesia. Caregivers reported that painful comorbidities could exacerbate dyskinesia; likewise, dyskinesia itself could cause pain. For example, Caregiver E described her child’s physical pain response: “
Overall, differentiating between pain caused by dyskinesia and pain caused by other sources was difficult. Caregivers often reported that pain and dyskinesia occurred concurrently, with dyskinesia also being triggered by a range of other cognitive/emotional, environmental, and personal factors.
Using their problem solving skills
Caregivers reportedly dealt with complex pain and dyskinesia presentations by using self-taught processes of elimination to confirm or negate possible sources of pain or dyskinesia until the triggering source was isolated. Caregiver C explained, “
However, the ability of caregivers to identify sources of pain and dyskinesia reportedly fluctuated given the inconsistent nature of presentations.
The challenge of training support staff
Caregivers perceived support staff (at school and home) to have varying ability to problem solve their child’s pain management. In many scenarios, caregivers felt the need to instruct support staff in the care needs of their child, despite staff having prior experience of working with their child. Caregivers found the prospect of training staff to be daunting due to the unique and complex nature of their child’s condition. “
Prioritization of pain communication
All caregivers reported they had learned to tune into their child’s individual way of communicating pain, including their distinctive physical and emotional responses, in order to identify pain.
In attempting to problem solve pain, caregivers prioritized actively engaging children with some capacity to self-report using augmentative and alternative communication (AAC). This was considered valuable even when only a portion of the information could be obtained (e.g., body location) or children’s responses were inconsistent or not easily interpreted.
However, the capacity to effectively communicate using AAC reportedly fluctuated depending on dyskinesia. Caregiver H explained, “
This theme represents the active approach caregivers took to identify solutions for their child’s chronic pain.
Motivation to self-learn
Caregivers reported they had developed the expertise to manage their child’s chronic pain over time through patterns of self-reflection and learning from past experiences.
Caregivers also sought to learn about successful ways to manage pain and dyskinesia from others with lived experience, with many connecting via internet chat forums. Caregiver D reported, “
Some caregivers also recognized the benefit in connecting with people with non-CP dystonia, because of their ability to report unbiased perspectives regarding their pain experiences.
Driven to advocate
Some caregivers reportedly adopted advocate roles in light of their child’s pain experience, often linked to feeling that health professionals overlooked their pain issues.
This theme relates to caregivers seeking greater fulfilment of their preferences for pain treatments within family-centred pain management, including nonpharmacological options.
Getting to the bottom of it
When dealing with their child’s pain, caregivers expressed a desire for health professionals to better diagnose and treat the underlying pain sources rather than symptomatically treating pain through prescribing pain medication.
Caregiver’s reluctance to use pain medications stemmed from past experiences of drug side effects such as fatigue and sedation, which they felt impacted their child’s quality of life and were difficult to detect in their child with communication limitations.
Seeking out nonpharmacological treatments
Many caregivers reported using a range of nonpharmacological treatments, with some exploring complementary (e.g., chiropractors, massage, mindfulness) and experimental (e.g., gut health diets) options. Caregiver H perceived such treatments to have a positive impact on pain reduction compared to alternatives from their experience.
Caregivers felt that allied health professionals offered limited additional pain interventions aside from providing postural management. Caregivers valued the role of allied health clinicians; however, they perceived them to “
This theme represents the emotional toll and psychosocial impacts on caregivers and families from supporting children through experiences of chronic pain.
The array of emotional impacts
Caregivers highlighted impacts to their emotional well-being including stress, anxiety, and low mood in response to supporting their child through the turbulent journey of chronic pain. Caregiver C emphasized the extent of her feelings following her child’s persisting pain following orthopaedic surgery: “
Influence over life roles
Caregivers acknowledged that managing pain in their child or adolescent often took precedence over other valued life roles and interests.
Some caregivers reported having restricted capacity to travel because of their child’s reduced sitting tolerance in response to chronic pain, which limited their ability to visit and socially interact with other friends and family members. As a result, some caregivers reported feeling socially isolated. Caregiver D explained, “
Caregiver H reflected on her experience of supporting her child with chronic pain, expressing that she felt like she had “
This theme relates to caregivers reflecting upon the trajectory of their child’s ongoing pain and dyskinesia throughout childhood.
Pain becoming entrenched in lives
Caregivers reflected that pain had become entrenched and a usual part of their child’s life as they developed.
Some caregivers reported that pain had progressed in their child.
The challenges of pain and dyskinesia with age
As children and adolescents aged, caregivers became more reliant on managing dyskinesia using equipment and external supports (e.g., wheelchairs, standing frames weighted blankets) compared to the physical strategies (e.g., hugging in a flexed position) used in younger children. Caregiver A reflected on her child as a baby:
However, with growth, she was now reliant on physical restraints to ensure her child’s safety, explaining, “
Discussion
This in-depth qualitative study illustrates how caring for children and adolescents with dyskinetic and mixed dyskinetic/spastic CP is a demanding and long term journey, with particular challenges faced because of their communication limitations. Caregivers highlighted that identifying and managing pain was complex, and they perceived gaps in health professionals’ skills to support pain management. Study findings provide valuable insight into the need for coordinated, interdisciplinary pain management in line with caregiver preferences that addresses practical needs such as formal education, access to nonpharmacological treatments, support for the emotional and physical well-being of caregivers, and timely equipment provision.
Previous qualitative research has explored caregiver perspectives of chronic pain management in severely impaired children with CP and other types of neurological impairment, but not dyskinetic CP [14, 38, 39]. Study findings resonate with themes previously identified in published research, including the central role of caregivers within pain communication, self-development of expertise, learning from past pain experiences, and the need to advocate for pain to be actively treated [38, 39]. Similarly, health professionals lack of prioritization of pain management amongst other medical needs and limitations in the efficacy of pain treatments available to children with CP has also been identified [14, 39, 40]. However, this study uncovered several themes/subthemes distinctive to children with CP and the complex overlay of dyskinesia as described below.
For caregivers, the inconsistent and unpredictable interactions between pain and dyskinesia added significant complexity to their daily caregiving routine. Other literature has recognized the range of extrinsic (e.g., equipment) and intrinsic (e.g., emotion, intent) influences over dyskinesia exacerbations including pain [2]. However, the lived experience of the complex bi-directional interactions between pain and dyskinesia, whereby dyskinesia can be both a pain source as well as a form of pain expression where other pain sources are present, have not been practically discussed in terms of their impact on pain management [9, 39]. In response to study findings, we posit that pain education should be early and routine and should incorporate advice about managing these complex pain and dyskinesia paradigms to support caregiver learning. Likewise, such education should underpin the training of support staff. Moving forward, a focus on basic education is essential, with education considered a lifespan priority for people with CP and the cornerstone of evidence based pain management in other chronic pain populations [41, 42]. A lack of basic pain education may compound issues of the under-detection and under-treatment of pain in CP populations and needs to be addressed in a targeted way for those with dyskinesia. By developing formal pain education and resources, consistent education can be disseminated across a range of key stakeholders for this unique population of children.
Caregivers felt that medical professionals relied on medication prescription and perceived limited access to additional nonpharmacological treatment options for pain. Children with dyskinetic CP may be particularly vulnerable to medication over-prescription due to their limited communication and the paucity of available evidence based nonpharmacological treatment options [24]. Overuse of pain medications is a common problem in other populations with chronic pain, leading to adverse patient side effects and significant economic burden [41]. Children and adolescents with CP are more likely to already be on a range of medications because of their multiple comorbid conditions and may be particularly predisposed to drug side effects secondary to polypharmacy [13]. For caregivers, medication concerns drove them to pursue non-pharmacological treatments shown to be cost effective and helpful for the longer term management of chronic pain in other populations [41]. Nonpharmacological treatments commonly used for children with CP may include exercise, aquatic therapy, thermotherapy, and massage [24, 43]. Passive (e.g., massage) therapies are often criticized for their short acting benefits in other populations with chronic pain and their lack of evidence base [44, 45]. However, despite limited evidence for massage, it is commonly used in this severely physically impaired population and may play a role where active therapies can be burdensome and difficult to access [19]. Overall, there is a need to explore the efficacy of nonpharmacological pain treatments alongside health professional knowledge and attitudes to pain management to better meet the needs of children and caregivers.
Caregivers were emotionally distressed and physically exhausted from supporting chronic pain needs in their children with high existing care demands. Such high care needs are likely to be further compounded by chronic pain, highlighting the vulnerable nature of this population of caregivers. Given that caregivers are already likely to have poor health and well-being directly linked to the high care needs of their children, it is crucial to provide a family-centred and bio-psychosocial approach to chronic pain management [22, 46]. Future consideration should be given to more targeted caregiver interventions, with other types of paediatric chronic pain populations starting to explore the efficacy of group programs that target pain education, effective communication, and development of coping skills within caregivers [47]. For caregivers of children with CP and dyskinesia, these interventions need to account for barriers and facilitators identified by this study (e.g., online accessibility).
The strength of this study lies in the exploration of real-world caregiver experiences of managing pain in children and adolescents with dyskinetic and mixed dyskinetic/spastic CP, having previously reported on the child experience [48]. Study weaknesses include the presence of reporting bias, with the impact of pain on children being reported through the lens of caregivers [49]. However, this study did not aim for caregivers to provide a direct proxy-report for their child’s pain, as this has been already reported in the authors’ broader body of work [48]. There may also be some contextual differences between caregiver experiences of chronic pain in younger children (5 to 12 years) compared to adolescents (13 to 18 years) and those with dyskinetic compared to mixed spastic/dyskinetic CP. However, given the small proportional representation of children with dyskinetic CP, it was appropriate to sample younger children and adolescents and the different classifications of dyskinesia together in this study. Other methodological limitations include limited piloting of the transcript and follow up of those who did not respond to member checking. However, parents of this complex population are generally time poor and we did not want to overburden them.
Future research needs to explore health professional and support staff perspectives of managing chronic pain in this population, so findings from this study can be triangulated and meaningful strategies developed to progress pain management. There may also be benefit in undertaking quantitative studies that explore the impact of dyskinesia severity and different types of muscle tone/movement disorder on pain presentations to help inform future diagnostic and management strategies in this population.
Conclusions
Caregivers faced significant challenges to effectively managing chronic pain in their children. There is a need for early structured pain education and resources for caregivers and support staff (at home and school) that address managing pain amongst the complex overlay of dyskinesia. There is further need to look towards strategies that ensure caregiver preferences for nonpharmacological treatments are met within pain management.
Key messages
There is a lack of formal pain education provided to caregivers and support workers.
Caregivers would like more access to nonpharmacological treatments for pain.
There is a need to look towards strategies to support caregiver well-being within pain management.
Footnotes
Acknowledgments
The authors would like to thank Ms. Catherine Clancy (Victorian Paediatric Rehabilitation Service, Monash Children’s Hospital), Ms. Mahek Dudhwala (Victorian Paediatric Rehabilitation Service, Monash Children’s Hospital), Associate Professor Michael Fahey (Victorian Paediatric Rehabilitation Service, Monash Children’s Hospital), Dr. Kate Willoughby (Royal Children’s Hospital), and Associate Professor Adam Scheinberg (Victorian Paediatric Rehabilitation Service, Royal Children’s Hospital) for their contributions and in-kind support of this study.
Clare McKinnon is funded to complete her PhD by the Australian Government Research Training Program Scholarship and Centre for Research Excellence in Cerebral Palsy, Murdoch Children’s Research Institute, Top-Up Scholarship. She also received a Career Development Grant from The Research Foundation, Cerebral Palsy Alliance 2017 which helped to fund this study. Adrienne Harvey receives a National Health and Medical Research Council Translating Research Into Practice Fellowship 2018. This project was supported by Murdoch Children’s Research Institute and the Victorian Governments Operational Infrastructure Support Program.
Conflict of interest
The authors have no conflict of interest to report.