DEVELOPMENTAL MEDICINE & CHILD NEUROLOGY
ORIGINAL ARTICLE
Impact of child and family characteristics on cerebral palsy treatment GIJA RACKAUSKAITE 1
| PETER W ULDALL 2 | BODIL H BECH 3 | JOHN R ØSTERGAARD 4
1 Department of Paediatrics, Aarhus University Hospital, Aarhus; 2 The Cerebral Palsy Registry, National Institute of Public Health, University of Southern Denmark, Copenhagen; 3 Department of Public Health, Aarhus University, Aarhus; 4 Department of Paediatrics, Aarhus University Hospital, Aarhus, Denmark. Correspondence to Gija Rackauskaite at Department of Paediatrics, Aarhus University Hospital, Brendstrupgaardsvej 100, 8200 Aarhus N, Denmark. E-mail: [email protected] This article is commented on by Bassan on pages 894–895 of this issue.
PUBLICATION DATA
Accepted for publication 30th March 2015. Published online 8th May 2015. ABBREVIATIONS
CP-TQ NCPR
Cerebral Palsy Treatment Questionnaire Danish National Cerebral Palsy Registry
AIM The aim of the study was to describe the relationship between the child’s and family’s characteristics and the most common treatment modalities in a national population-based sample of 8- to 15-year-old children with cerebral palsy. METHOD A cross-sectional study, based on the Danish Cerebral Palsy Registry. The parents of 462 children answered a questionnaire about their child’s treatment and the family’s characteristics (living with a single parent, having siblings, living in a city, parental education level). Descriptive and logistic regression analyses were performed for every treatment modality, stratified by Gross Motor Function Classification System (GMFCS) level. RESULTS An IQ below 85 was associated with weekly therapy in GMFCS level I (adjusted odds ratio [ORadj] 2.5 [CI 1.1–5.7]) and the use of oral spasmolytics in GMFCS levels III to V (ORadj 3.1 [CI 1.3–7.4]). Older children in GMFCS levels III to V used daily orthoses less frequently (ORadj 0.7 [CI 0.6–0.9] per year). Of all of the family characteristics studied, only the parents’ education level had significant associations with more than one treatment modality. INTERPRETATION A child’s cognitive function showed an impact on treatment of the motor impairment in children 8 to 15 years of age with cerebral palsy. Parental education level may influence the choice of treatment.
Children and adolescents with cerebral palsy (CP) receive a wide range of treatment modalities for both motor disability and associated comorbidity. The treatment of motor disability includes rehabilitation services such as physiotherapy, occupational and speech therapy, orthotic devices, orthopaedic surgery, and oral, intramuscular or intrathecal medication. Unfortunately, evidence in support of many of the treatment modalities is lacking;1 therefore, factors other than the clinical features may determine the treatment of CP. For example, a significant geographical variation was revealed in the parent-reported unmet needs in the care of children with severe disability, including CP,2 and the social functioning of children with CP has been reported to be associated with family characteristics such as a higher parental education level.3 It is unknown whether compliance with treatment is also associated with family characteristics. In a study of paediatric traumatic brain injury,4 it was shown that environmental factors were significant predictors of the child’s functioning and participation. We hypothesized that the family’s characteristics, such as parental education level, living with a single parent, or having siblings, and the child’s characteristics (age, sex, cognitive impairment) might have an impact on the treatment given to children with CP beyond the severity of their motor impairment. The aim of the present study is to 948 DOI: 10.1111/dmcn.12791
describe the relationship between child and family characteristics and treatment modalities, such as physiotherapy, occupational therapy, orthoses, orthopaedic surgery, oral spasmolytic medication, and botulinum toxin (BoNT-A) injections, in a national population-based sample of 8- to 15-year-old children according to their motor impairment level.
METHOD A cross-sectional national study based on the Danish National Cerebral Palsy Registry (NCPR) and a questionnaire administered to the parents of 8- to 15-year-old children with CP. Participants Children (n=977) with CP who were born between 1997 and 2003 were identified from the Danish NCPR. Inclusion criteria for the NCPR were a child born in Denmark and a diagnosis of congenital CP confirmed at the age of 4 to 5 years by a neuropaediatrician.5 The register was validated through systematic cross-reference with the Danish National Patient Register, which automatically registered all diagnoses given in the health care system in Denmark.6 The civil statuses and addresses of the child were updated from the national Civil Registration System in © 2015 Mac Keith Press
September 2011. Children who emigrated from Denmark or died before September 2011 or during the study period were excluded (n=79). Further, two children could not be found at the address. In addition, in two children, the CP diagnosis could not be confirmed, and one had a slowly progressive disease. A total of 893 children were thus eligible for the study. The invitation letter was addressed to the parents from January 2012 (pilot-testing) to January 2013 and included login information for an online questionnaire. A reminder was sent after 1 month, and a second, including the questionnaire in paper form, was sent after 2 months. The study was approved by the Danish Data Protection Agency (reference number 2008-41-2927) and the Ethical Committee of Central Denmark Region (reference number M-20090016).
Data sources Because a valid description of the frequency of the occupational therapy and physiotherapy seldom is available in the medical records, a parental CP Treatment Questionnaire (CP-TQ) was developed. The questionnaire was developed by an expert group, which included neuropaediatricians, physiotherapists, and an orthopaedic surgeon. The CP-TQ was then adjusted to comments from two parents and pilot-tested on 23 families who had a child with CP. The parental answers were consistent with the medical records from Aarhus University Hospital, where information about all treatment modalities was recorded. These 23 children were included into the study because no major adjustments were made to the CP-TQ after that stage. The CP-TQ contained information about physiotherapy, occupational therapy, uses of orthoses, surgery, prescribed groups of medications, and BoNT-A injections. In addition, questions about family and school characteristics were included. An English version of the CP-TQ questions included in this study is provided in Appendix S1 (online supporting information). The frequency of therapy was defined as the frequency of physiotherapy and occupational therapy provided by the therapist within the previous 12 months. If parents stated that the child received one type of therapy weekly and another type of therapy less than weekly, the total frequency of therapy was defined to be more than weekly. If both therapies were stated as ‘two to three times per month’, the total frequency was assigned as weekly. The duration of therapy was not reported because we assumed it to be fluctuating and therefore difficult to report retrospectively. Usually, such sessions lasted about 30 minutes. In terms of orthoses, only daily or nightly used orthoses were included as an outcome variable. Spasmolytics were the most common used oral medications and were therefore included as an outcome variable. Orthopaedic surgery included any ‘operation in the legs, arms or spine due to cerebral palsy ever performed’. It did not include other operations such as intrathecal baclofen pump and gastrostomy.
• • •
What this paper adds An increased frequency of therapy and interventions was observed in higher Gross Motor Function Classification System levels in children with cerebral palsy. The child’s cognitive ability had an impact on the choice of treatment for motor disability. Parental education level may influence the use of certain treatment modalities.
We included only intramuscular BoNT-A injections ever received as an outcome variable. The date of birth and sex were available in the NCPR. For the non-respondents, the age of the child was calculated at the date of the deadline for the first reminder. The child’s site of living and region were available from the Civil Registration System. The number of parents (single/two), number of siblings (0 or ≥1), and education level of the parents were retrieved from the CP-TQ. Parental education was summarized from an original seven levels into three levels: short, middle (3–4y of education after graduating high school), and high (at least 5y of education after graduating high school). In the case of parents having different educational levels, the highest education level was used. The type of CP and developmental quotient were extracted from the NCPR, where the cognitive development was categorized into three levels: IQ>85, IQ 85 to 50, and IQ85 IQ 50–85 IQ
ORIGINAL ARTICLE
Impact of child and family characteristics on cerebral palsy treatment GIJA RACKAUSKAITE 1
| PETER W ULDALL 2 | BODIL H BECH 3 | JOHN R ØSTERGAARD 4
1 Department of Paediatrics, Aarhus University Hospital, Aarhus; 2 The Cerebral Palsy Registry, National Institute of Public Health, University of Southern Denmark, Copenhagen; 3 Department of Public Health, Aarhus University, Aarhus; 4 Department of Paediatrics, Aarhus University Hospital, Aarhus, Denmark. Correspondence to Gija Rackauskaite at Department of Paediatrics, Aarhus University Hospital, Brendstrupgaardsvej 100, 8200 Aarhus N, Denmark. E-mail: [email protected] This article is commented on by Bassan on pages 894–895 of this issue.
PUBLICATION DATA
Accepted for publication 30th March 2015. Published online 8th May 2015. ABBREVIATIONS
CP-TQ NCPR
Cerebral Palsy Treatment Questionnaire Danish National Cerebral Palsy Registry
AIM The aim of the study was to describe the relationship between the child’s and family’s characteristics and the most common treatment modalities in a national population-based sample of 8- to 15-year-old children with cerebral palsy. METHOD A cross-sectional study, based on the Danish Cerebral Palsy Registry. The parents of 462 children answered a questionnaire about their child’s treatment and the family’s characteristics (living with a single parent, having siblings, living in a city, parental education level). Descriptive and logistic regression analyses were performed for every treatment modality, stratified by Gross Motor Function Classification System (GMFCS) level. RESULTS An IQ below 85 was associated with weekly therapy in GMFCS level I (adjusted odds ratio [ORadj] 2.5 [CI 1.1–5.7]) and the use of oral spasmolytics in GMFCS levels III to V (ORadj 3.1 [CI 1.3–7.4]). Older children in GMFCS levels III to V used daily orthoses less frequently (ORadj 0.7 [CI 0.6–0.9] per year). Of all of the family characteristics studied, only the parents’ education level had significant associations with more than one treatment modality. INTERPRETATION A child’s cognitive function showed an impact on treatment of the motor impairment in children 8 to 15 years of age with cerebral palsy. Parental education level may influence the choice of treatment.
Children and adolescents with cerebral palsy (CP) receive a wide range of treatment modalities for both motor disability and associated comorbidity. The treatment of motor disability includes rehabilitation services such as physiotherapy, occupational and speech therapy, orthotic devices, orthopaedic surgery, and oral, intramuscular or intrathecal medication. Unfortunately, evidence in support of many of the treatment modalities is lacking;1 therefore, factors other than the clinical features may determine the treatment of CP. For example, a significant geographical variation was revealed in the parent-reported unmet needs in the care of children with severe disability, including CP,2 and the social functioning of children with CP has been reported to be associated with family characteristics such as a higher parental education level.3 It is unknown whether compliance with treatment is also associated with family characteristics. In a study of paediatric traumatic brain injury,4 it was shown that environmental factors were significant predictors of the child’s functioning and participation. We hypothesized that the family’s characteristics, such as parental education level, living with a single parent, or having siblings, and the child’s characteristics (age, sex, cognitive impairment) might have an impact on the treatment given to children with CP beyond the severity of their motor impairment. The aim of the present study is to 948 DOI: 10.1111/dmcn.12791
describe the relationship between child and family characteristics and treatment modalities, such as physiotherapy, occupational therapy, orthoses, orthopaedic surgery, oral spasmolytic medication, and botulinum toxin (BoNT-A) injections, in a national population-based sample of 8- to 15-year-old children according to their motor impairment level.
METHOD A cross-sectional national study based on the Danish National Cerebral Palsy Registry (NCPR) and a questionnaire administered to the parents of 8- to 15-year-old children with CP. Participants Children (n=977) with CP who were born between 1997 and 2003 were identified from the Danish NCPR. Inclusion criteria for the NCPR were a child born in Denmark and a diagnosis of congenital CP confirmed at the age of 4 to 5 years by a neuropaediatrician.5 The register was validated through systematic cross-reference with the Danish National Patient Register, which automatically registered all diagnoses given in the health care system in Denmark.6 The civil statuses and addresses of the child were updated from the national Civil Registration System in © 2015 Mac Keith Press
September 2011. Children who emigrated from Denmark or died before September 2011 or during the study period were excluded (n=79). Further, two children could not be found at the address. In addition, in two children, the CP diagnosis could not be confirmed, and one had a slowly progressive disease. A total of 893 children were thus eligible for the study. The invitation letter was addressed to the parents from January 2012 (pilot-testing) to January 2013 and included login information for an online questionnaire. A reminder was sent after 1 month, and a second, including the questionnaire in paper form, was sent after 2 months. The study was approved by the Danish Data Protection Agency (reference number 2008-41-2927) and the Ethical Committee of Central Denmark Region (reference number M-20090016).
Data sources Because a valid description of the frequency of the occupational therapy and physiotherapy seldom is available in the medical records, a parental CP Treatment Questionnaire (CP-TQ) was developed. The questionnaire was developed by an expert group, which included neuropaediatricians, physiotherapists, and an orthopaedic surgeon. The CP-TQ was then adjusted to comments from two parents and pilot-tested on 23 families who had a child with CP. The parental answers were consistent with the medical records from Aarhus University Hospital, where information about all treatment modalities was recorded. These 23 children were included into the study because no major adjustments were made to the CP-TQ after that stage. The CP-TQ contained information about physiotherapy, occupational therapy, uses of orthoses, surgery, prescribed groups of medications, and BoNT-A injections. In addition, questions about family and school characteristics were included. An English version of the CP-TQ questions included in this study is provided in Appendix S1 (online supporting information). The frequency of therapy was defined as the frequency of physiotherapy and occupational therapy provided by the therapist within the previous 12 months. If parents stated that the child received one type of therapy weekly and another type of therapy less than weekly, the total frequency of therapy was defined to be more than weekly. If both therapies were stated as ‘two to three times per month’, the total frequency was assigned as weekly. The duration of therapy was not reported because we assumed it to be fluctuating and therefore difficult to report retrospectively. Usually, such sessions lasted about 30 minutes. In terms of orthoses, only daily or nightly used orthoses were included as an outcome variable. Spasmolytics were the most common used oral medications and were therefore included as an outcome variable. Orthopaedic surgery included any ‘operation in the legs, arms or spine due to cerebral palsy ever performed’. It did not include other operations such as intrathecal baclofen pump and gastrostomy.
• • •
What this paper adds An increased frequency of therapy and interventions was observed in higher Gross Motor Function Classification System levels in children with cerebral palsy. The child’s cognitive ability had an impact on the choice of treatment for motor disability. Parental education level may influence the use of certain treatment modalities.
We included only intramuscular BoNT-A injections ever received as an outcome variable. The date of birth and sex were available in the NCPR. For the non-respondents, the age of the child was calculated at the date of the deadline for the first reminder. The child’s site of living and region were available from the Civil Registration System. The number of parents (single/two), number of siblings (0 or ≥1), and education level of the parents were retrieved from the CP-TQ. Parental education was summarized from an original seven levels into three levels: short, middle (3–4y of education after graduating high school), and high (at least 5y of education after graduating high school). In the case of parents having different educational levels, the highest education level was used. The type of CP and developmental quotient were extracted from the NCPR, where the cognitive development was categorized into three levels: IQ>85, IQ 85 to 50, and IQ85 IQ 50–85 IQ
