Background and aim. Children and adolescents with cerebral palsy (CP) are at high risk
for pain and their pain is frequently underestimated and undermanaged. They often
experience a substantial pain burden from the pain itself and its influence on all
aspects of their life, for example ability to perform daily activities, participate
socially, sleep, or on overall quality of life. The overarching aim of the CPPain-program
is to reduce the pain burden in children and adolescents living with CP.
Study design. The CPPain has a prospective cohort comparative design and will include
before- and after measurements and process evaluation of a nested intervention. This
registration concerns the CPPain baseline and includes a multinational survey (the CPPain
survey) and qualitative interviews with children and adolescents with CP, their
parents/caregivers and health care professionals. The nested intervention will later be
developed based on findings from the baseline.
Qualitative data collection (the CPPain survey). The study population for the CPPain
survey will include children younger than 18 years with a CP diagnosis (=primary
participants) and their siblings (12-17 years) as controls. Parents of all primary
participants will provide proxy report. Children with CP > 8 years of age and cognitively
able (approx. 50%) and siblings (12-17 years) will provide self-report. The survey will
include a set of previously published and validated measures that assess aspects of pain
(experience, interference and management), performance of daily activities, mental
health, Health-Related Quality of Life (HRQoL), and participation. These measures include
modified versions of Dalhousie Pain Interview (DPI) and Brief Pain Inventory (BPI), Pain
Catastrophizing Scale (PCS), The Caregiver Priorities and Child Health Index of Life with
Disabilities (CPCHILD), Revised Children's Anxiety and Depression Scale (RCADS-25),
KidScreen, and Child and Adolescent Scale of Participation (CASP). We have also added a
researcher-developed questionnaire on COVID-19 and pain. We will have three versions of
the survey: Parent proxy, Self-report - full (12-18), and self-report - abbreviated (8-12
and older children with cognitive limitations). In addition to self- and/or
proxy-reported survey data, demographic and disability-related data already registered in
either national registers or electronic medical charts will be collected. Survey data
will primarily be collected using an electronic survey. The Clinical Data Management
System (CDMS) used for the eCRF in this study is Viedoc. The setup of the study specific
eCRF in Viedoc will be performed by Research Support Services CTU, Oslo University
Hospital (OUS).
Qualitative data collection. Potential participants for the child and parent interviews
will be identified from the CPPain survey or clinicians in the child habilitation
services. Inclusion criteria are school-aged children and adolescents (8-18 years) with
cerebral palsy who have pain, determined from their or their parents' responses to the
modified Dalhousie Pain Interview. If the child is unable to self-report or do not wish
to participate only the parent/caregiver will be included.A purposeful sampling strategy
will be used to ensure a diverse sample concerning child and parent age and gender, as
well as the child's level of physical and cognitive disability and severity of pain
burden. We estimate that we will include 15-20 child/parent dyads, but the final number
will depend upon the richness of the material and data saturation.Both individual
semi-structured interviews and focus groups will be used to answer the research
objectives. Interview guides cover experiences with pain and its impact on life and
living for children and parents respectively, parents' perception of their child's pain
burden and its impact, as well as how pain is managed, including a detailed mapping of
both the current and ideal help network. From the existing help network, identified by
interview participants, health care professionals will be identified and recruited for a
second round of interviews.
Data analysis. Quantitative questionnaire and register data will be analyzed using
appropriate statistical methods for comparing groups and analyzing time trends in close
cooperation with the study's statistician. Stratification will be used to investigate sub
groups. Analysis will be adjusted for appropriate sociodemographic variables. SPSS will
be the main tool for statistical analysis, while some of the more complex analysis will
require the use of R. Other specialized software will be applied as needed. Qualitative
data from interviews and from unstructured responses to survey questions will be analyzed
using appropriate qualitative methods; primarily content, thematic and/or network
analysis. NVivo will be the main qualitative analysis tool. Qualitative and quantitative
data will be combined and analyzed from either a quantitative or qualitative perspective
to provide a more comprehensive understanding of the phenomenon.