Health-related quality of life and functional outcome measures for children with cerebral palsy

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1 Health-related quality of life and functional outcome measures for children with cerebral palsy Jane W Schneider* PhD PT, Northwestern University Medical School, Physical Therapy and Human Movement Sciences, Children s Memorial Hospital, Chicago; Linda M Gurucharri MPT, Louis A Weiss Memorial Hospital, Chicago; Allison L Gutierrez MPT, Early Intervention Center of Brooklyn, Brooklyn New York, NY; Deborah J Gaebler-Spira MD, Rehabilitation Institute of Chicago, Northwestern University Medical School, Department of Rehabilitation Medicine, Chicago IL, USA. *Correspondence to first author at Northwestern University Medical School, Physical Therapy and Human Movement Sciences, 645 N Michigan Ave, Suite 1100 Chicago, IL 60611, USA. j-schneider@northwestern.edu The aim of this study was to examine measures of healthrelated quality of life (HRQL) in children with cerebral palsy (CP) by comparing scores of a generic HRQL measure, the Child Health Questionnaire (CHQ); a disease-specific HRQL measure for children with CP, the Caregiver Questionnaire (CQ); and a pediatric functional measure, the Wee- Functional Independence Measure (WeeFIM ). Participants included 30 caregivers of children with CP. The caregivers children were a mean age of 8 years 6 months (17 females, 13 males). The ethnic origin of the children was 18 African- American, 8 white, 3 Hispanic, and 1 Middle Eastern. Significant correlations were found between the CQ and WeeFIM total and subscale scores (r=0.388 to 0.641). There was no correlation between the CHQ and CQ total summary scores, but significant correlations were found between the CHQ subscales related to parent time and family cohesion and the CQ total and subscale scores (r=0.386 to 0.481). The lack of correlation between the CHQ and WeeFIM indicates HRQL and function are different constructs that cannot be inferred from each other. The fair relationship found between the CQ and WeeFIM suggests that the constructs measured in these two assessments overlap. The lack of correlation between the total summary scores of the CHQ and CQ suggests the CQ may be a more specific measure of HRQL for this population that reflects the impact of the child s condition on the caregiver. Within the past 20 years three basic constructs for assessment tools have emerged (Haley 1994). Initially, the developmental framework of assessment dominated. This framework was based on the belief that interventions should be directed at helping children attain sequential developmental positions and move normally within them (Nelson 1995). These assessments were abundant in their descriptive nature, and their explanations of abnormality decidedly shaped therapeutic practice (Haley 1994). Clinicians, however, realized that assessment needed to be tied more closely to function rather than evaluating the child s ability to attain motor milestones. Thus, the focus of assessment moved to measuring function and the ability to complete activities of daily living at home and in the community (Haley 1991, 1992; McAuliffe et al. 1998). Recently, children s feelings about their ability to fulfill childhood roles have begun to be included in assessment even in those children with milder developmental impairments (Feldman et al. 2000, Saigal 2000, Sheridan et al. 2000). This construct, known as health-related quality of life (HRQL), measures how people feel about aspects of their lives directly related to health, excluding issues such as religious beliefs and practices (Guyatt et al. 1997, McLaughlin and Bjornson 1998). It is difficult, however, to measure quality of life due to widely divergent conceptualizations of a good life and what constitutes health (Dijkers 1999). For example, while two children may have the same functional limitations it cannot be assumed that they will experience the same quality of life. In general, HRQL measures aim to provide a more complete picture of the individual that is complementary to specific functional assessments, the traditional focus of clinicians (Rosenbaum and Saigal 1996). Besides their descriptive value, HRQL measures can also evaluate the effect of interventions on overall well-being (Guyatt et al. 1997). Measures of HRQL may be used to predict the future status of persons with an illness or condition (Rosenbaum and Saigal 1996). Thus, an HRQL measure used for a child with cerebral palsy (CP) may be a better indicator of social emotional adaptation in school than a functional assessment measure (Rosenbaum and Saigal 1996). There are two types of HRQL assessment instruments: generic and disease-specific. Generic instruments are designed to be applicable to all human populations (Dijkers 1999). As such, generic tools can be used for comparing HRQL across populations of people with different conditions (Guyatt et al. 1997). While a generic instrument may be generalizable, it will provide a more superficial evaluation of a person and will be of limited value in identifying the specific effects of a disease process (Dijkers 1999). Conversely, disease-specific measures may provide a more clinically relevant HRQL evaluation directed to the symptoms, problems, or side effects of a certain condition (Guyatt et al. 1997, Rutishauser et al. 1998, Dijkers 1999). For example, HRQL assessment for children with CP might include questions related to the effects of spasticity while a generic tool will not address this issue. The disease-specific instruments are more powerful in detecting treatment effects and more sensitive to small changes in (sub-) domains of particular concern to clinicians or patients (Dijkers 1999). Currently, generic instruments have been well developed and fine-tuned with many years of research (Dijkers 1999). Unfortunately, disease-specific instruments have not been developed to the same extent and are often either unavailable or developed solely for a particular study (Dijkers 1999, 2000). Developmental Medicine & Child Neurology 2001, 43:

2 There is a need for HRQL measures that are standardized, valid, reliable, inexpensive, and provide a quick and easy means of measuring quality of life (Dijkers 1999, 2000; Hack 1999). More specifically, there is an immediate demand for research on HRQL instruments applicable to children, especially those with disabilities. A cumulative review of HRQL measurement tools found less than 5% of published HRQL measures to be applicable to children and even fewer were applicable to children with developmental disabilities (Berzon 1993). The small number of HRQL assessment tools, however, is not indicative of their need. As an illustration, the United Cerebral Palsy Association places quality of life among their highest priorities (McLaughlin and Bjornson 1998). Moreover, the issues addressed in HRQL measures are often the foremost concerns identified by children and parents and should become part of routine clinical assessments (Haley 1994). Accordingly, major funding sources now expect quality of life to be one of the clinical research outcome measures (McLaughlin and Bjornson 1998). Valid and reliable HRQL assessment tools that are sensitive to detecting change are needed to meet this present demand (Dijkers 1999). Several methodological issues related to reliability and validity must be considered when developing HRQL measures for children with disabilities. The issue of cognitive and developmental limitations arises when assessing HRQL in children. Quality of life measures tend to be self-administered surveys, yet children may not be capable of answering abstract questions independently. Parents are often relied upon as proxies to assess children s levels of disability. Although parents do not always concur on all aspects of the child s HRQL, they are more reliable than other proxies such as teachers and health professionals (Rosenbaum and Saigal 1996). Landgraf and coworkers (1996, 1998) found relatively high agreement between mothers and their children for certain concepts of HRQL. After a research review, Rosenbaum and Saigal (1996) concluded that parents serve as an excellent proxy for their children when assessing HRQL, despite the potential for some degree of distortion. The purpose of this study was to investigate the relationships between the scores of a generic pediatric HRQL assessment, the Child Health Questionnaire (CHQ; Landgraf et al. 1996); a disease-specific pediatric HRQL assessment tool, the Caregiver Questionnaire (CQ); and a functional outcome measure, the Wee-Functional Independence Measure (WeeFIM; WeeFIM SystemSM 1998). The CHQ is a generic HRQL tool that measures the physical and psychosocial well-being of children with or without a disability above the age of 5 years. The CHQ is a comprehensive tool that consists of 14 basic content domains representing the most essential components of a child s HRQL (Landgraf et al. 1996). Four versions of the CHQ currently exist. Three of the versions, the CHQ- PF98, PF50, and PF28, are parent-completed questionnaires that differ only in length. The fourth version, the CHQ-CF87, is a child-completed questionnaire. The CHQ assessment tool is the product of the Child Health Assessment Project initiated in 1990 to advance methods of studying HRQL in children across diverse populations (Landgraf et al. 1998). Throughout its development, crosscultural evaluation as well as standardized analytic methods were used to ensure the CHQ s validity and reliability (Landgraf et al. 1996, 1998). The CHQ s validity as a generic pediatric HRQL measure was studied by experts in 11 countries/languages during the CHQ s construction and by comparing the CHQ s content domains to seven other published child health assessment measures (Landgraf et al. 1996). Results showed that the CHQ was able to distinguish between children with and without disabilities including asthma, attention-deficit hyperactivity disorder, juvenile rheumatoid arthritis, and epilepsy (Landgraf et al. 1996). The average success rate of item discriminant validity for all the items of the CHQ-PF28, the shorter parent-completed version used in this study, was 94% and all items exceeded the minimum criteria for item internal consistency by an average of 94% (Landgraf et al. 1996). The CQ is a disease-specific measure that was developed in 1990 at the Rehabilitation Institute of Chicago, USA for a defined population of children with spastic quadriplegic CP who were to undergo selective posterior rhizotomy. The items for the CQ were selected and developed by health care professionals (nurses, physicians, physical and occupational therapists), family members, and caregivers in response to therapeutic goal setting. Its purpose was to be a quick and easy HRQL measure used in conjunction with other more functionally-based outcome measures to assess the effect of interventions. The CQ measures the caregiver s difficulties and satisfaction with their child s progress in four areas: Personal Care, Positioning/Transferring, Comfort, and Interaction/ Communication. The original CQ has been recently modified to include items more appropriate for the general pediatric population with CP. The CQ has been presented internationally (Tann 1998, Revivo 2000) and used clinically to gather qualitative information about the effect of interventions. However, it has never been tested as a valid measure of quality of life for children with CP. In contrast to the two HRQL measures, the WeeFIM is a functional outcome instrument designed to measure restrictions or inability to perform self-care, mobility, and communication skills within the range considered normal (Msall et al. 1994a, b). Developed by the Task Force to Develop a Uniform Data System and based on the Functional Independence Measure (FIM) for adults, the WeeFIM measures the amount of assistance a child needs to perform routine daily activities (WeeFIM SystemSM 1998). The WeeFIM is appropriate for use with children without disabilities from 6 months to 8 years; children with developmental disabilities (DD) from 6 months to 15 years; and persons of all ages with DD and developmental ages less than 7 years (WeeFIM SystemSM 1998). This instrument provides a uniform rating scale and common language for describing levels of disability in children (WeeFIM SystemSM 1998). Unlike other commonly used pediatric functional assessments, the WeeFIM is geared to both health and developmental professionals to assess and track neurodevelopmental disabilities as well as evaluate the outcomes of biomedical, developmental, and psychosocial interventions (Msall et al. 1994a, 1997). Normative data for the WeeFIM were gathered in a largescale study on children without disabilities (Msall et al. 1994a). Studies have shown the WeeFIM to be a valid and reliable measure of disability in children with developmental disorders (Msall et al. 1990, 1991). Also, pilot data on the WeeFIM with children with CP, limb deficiency, Down syndrome, spina bifida, and extreme prematurity demonstrate that the WeeFIM is a valid measure for tracking disability during preschool and 602 Developmental Medicine & Child Neurology 2001, 43:

3 middle childhood (Msall et al. 1994b). The WeeFIM has test retest reliability (r=0.99), interrater reliability (r=0.95), and content validity for the amount of help (r=0.95) and time (r=0.88) necessary to complete tasks (Msall et al. 1990). In addition to being valid and reliable, a recent study also demonstrated that the WeeFIM is sensitive to measuring change in children with CP (McAuliffe et al. 1998). There were two main aims of the present study. The first was to investigate the relationship between the two HRQL measures: the CQ and the CHQ. If the CQ correlated with the CHQ, an established HRQL measure, this would help establish the validity of the CQ as a measure of HRQL in children with CP. The second aim of this study was to investigate the relationship between HRQL measures and a functional outcome measure, the WeeFIM. A strong positive relationship would suggest that using both types of measures might be redundant when assessing children with CP. Should no significant correlation exist then quality of life might not be inferred from functional ability. Health-related quality of life measures, therefore, should be included in clinical practice if improving quality of life is a major goal of treatment. It was hypothesized that the scores on the CQ would correlate significantly with the scores on the CHQ. Second, a non-significant correlation between the HRQL measures and the functional outcome measure was expected. Method PARTICIPANTS The 30 participants represented a convenience sample recruited from the CP clinic and outpatient pediatric physical therapy department at the Rehabilitation Institute of Chicago. Only one caregiver declined participation. Participants were the parent or primary caregiver of a child diagnosed with CP. The degree of the child s involvement ranged from mild to severe, however, the majority of the caregivers children (n=22) were estimated to be at the Gross Motor Functional Classification System (GMFC) III or IV (Palisano et al. 1997) indicating moderate disability (Table I). Caregivers children were between the ages of 5 and 15 years with a mean age of 8 years 6 months, 57% female, were predominately African-American (n=18), attended the clinic primarily for spasticity management, and were typical of the clinic population. Caregivers included mothers (n=19), fathers (n=5), grandmothers (n=2), foster parents (n=2), and others (n=2). The caregiver must have lived with the child for the 6 months before participation in the study. Participants were fluent in the English language and signed an informed consent document before participating in the study. MEASUREMENTS The generic CHQ is designed to measure the physical and psychosocial well-being of children above 5 years of age, Table I: Description of children of participants Child Age (y:m) Sex Race GMFC Reason for clinic visit 1 7:11 F AA III SM 2 10:4 M AA III SM 3 10:2 M AA III SM 4 10:2 F AA III SM 5 6:0 F W IV SM/OF 6 7:2 M AA V OF 7 8:9 F ME III OF 8 7:3 F AA IV OF/SM 9 5:11 F AA I SM 10 7:7 M W IV SM 11 8:1 F W V SM 12 11:0 F AA III SM 13 10:5 F AA V SM 14 5:7 F HIS V SM 15 7:2 F HIS III SM 16 6:0 M W IV SM 17 6:9 M AA IV SM/OF 18 5:8 M W I SM 19 10:0 F W IV PT 20 15:1 M AA IV OF 21 6:3 F AA III OF 22 8:0 M HIS III SM 23 15:4 M W I SM 24 9:8 F AA IV SM 25 7:6 M W IV SM 26 5:3 F AA IV SM 27 7:3 F AA IV SM 28 7:1 M AA III OF 29 8:4 F AA IV OF 30 14:5 M AA II OF AA, African-American; W, White; ME, Middle Eastern; HIS, Hispanic; SM, Spasticity management; OF, Othopedic follow-up; PT, Physical therapy only; GMFC, Gross Motor Function Classification (levels I to V). Health-Related Quality of Life and Functional Outcome Measures in CP Jane W Schneider et al. 603

4 regardless of the presence or type of disability (Landgraf et al. 1996). The version used in this study, the CHQ-PF28, is a 28-item caregiver-completed questionnaire that asks questions about the child in 10 health-related quality of life domains. These domains include physical functioning, limitations in school and activities due to physical health, limitations due to emotional health, limitations due to behavior issues, general health perceptions, bodily pain, self-esteem, mental health, general behavior, and changes in health (Landgraf et al. 1996). The CHQ-PF28 also includes four additional domains that ask questions about the impact of the child s health status on the caregiver and family unit. The CHQ subscales of Parental Impact Time (CHQ-PT) and Parental Impact Emotional (CHQ-PE) represent the impact of the child s health on the parent s time and emotional state. The CHQ subscales of Family Impact Activities (CHQ- FA) and Family Cohesion (CHQ-FC) represent the impact of the child s health on family activities and the overall cohesion of the family unit (Landgraf et al. 1996). The scoring procedure for the CHQ used in this study was consistent with the procedure described in the CHQ User Manual and is summarized below (Landgraf et al. 1996). The CHQ raw score for each question is converted into a 0 to 100 scale with higher scores reflecting better well-being (Landgraf et al. 1996). The 10 domains of the CHQ are not summed to derive one total score, but rather two different summary scores, a physical summary score and a psychosocial summary score. Calculations of summary scores are based on normative data from the general population of the USA, and a weighting factor is applied for each subscale (Landgraf et al. 1996). The CHQ physical summary score is weighted to represent principally the subscales of physical functioning, role limitations due to physical health, general health, and bodily pain. The CHQ psychosocial summary score is weighted to represent principally the subscales of self-esteem, mental health, general behavior, and role limitations due to emotional health and behavioral issues. The disease-specific HRQL measure, the CQ, is a 29-item self-administered questionnaire that consists of four sections: (1) Personal Care, (2) Positioning/Transferring, (3) Comfort, and (4) Interaction. Caregivers rate each task by making a mark on an unnumbered 10-centimeter line that corresponds to the degree of difficulty (Appendix). To score the CQ, each mark on each 10-centimeter line is transformed into a linear analog scale in which a score of zero reflects maximal ease and 10 reflects maximum difficulty. For this study, one question in the Comfort section was omitted from the scoring since the answers to the question could not be interpreted as reflecting either high or low levels of comfort. The remaining 28 questions from the four CQ sections were computed into a total summary score. The first three sections of the CQ were combined to represent a Physical subscale in order to make comparisons with the Physical summary score of the CHQ. The fourth section of the CQ, the Interaction section, was compared with the CHQ Psychosocial summary score. The CQ scores were calculated by finding the arithmetic mean of the questions included in each of the categories. If a question was marked not applicable, it was omitted from the calculations. The WeeFIM is an 18-item functional outcome measure that assesses a child s need for assistance to perform basic daily tasks on a routine basis (WeeFIM SystemSM 1998, McAuliffe et al. 1998). Each item is rated on a scale from complete dependence (score of 1) to complete independence (score of 7). The 18 functional tasks are grouped into three domains of self-care, mobility, and cognition. The assessment was administered in this study by interviewing the primary caregiver. A total raw score was calculated on the WeeFIM, as well as raw scores for the three general domains of self-care, mobility, and cognition (WeeFIM SystemSM 1998). Scoring procedures were consistent with the procedure outlined by the WeeFIM training manual (WeeFIM SystemSM 1998). Due to the fact that the WeeFIM is administered via interview, to ensure reliability the two investigators took part in a training program developed by the WeeFIM authors at Children s Hospital of Buffalo, Center for Functional Assessment Research (WeeFIM SystemSM 1998). Each investigator was certified by the Center to administer the WeeFIM reliably. In addition, the investigators demonstrated interrater reliability on 10 WeeFIM preliminary parent interviews with an intraclass correlation coefficient (ICC) value of at least 0.98 for all subscale and total score correlations. The ICC, originally designed for use with interval or ratio level data, has been shown to be applicable to ordinal level data without distortion when intervals between scores are assumed to be equal (Portney and Watkins 1993, Ottenbacher et al. 1996, Sperle et al. 1997). Based on the WeeFIM Training Protocol and the ICC interrater reliability data, both raters were considered reliable and qualified administrators of the WeeFIM. PROCEDURE Participants completed the three different health assessment tools while waiting for their child s appointments at the CP clinic or the outpatient pediatric physical therapy department of the Rehabilitation Institute of Chicago. As the CHQ and CQ were expected to contain similar content, the order of CHQ and CQ administration was alternated between the first or third assessment in order to minimize test order bias. The three assessments were administered by one of two student physical therapists after the assessments had been explained and written instructions were reviewed. The investigator was also available to answer questions throughout the testing. The total time to complete the three assessment tools was approximately 30 to 40 minutes. STATISTICAL ANALYSIS A two-tailed analysis using the Spearman s rank correlation coefficient (r) determined if a correlation existed between the total scores as well as the various subscales of the three different assessments for each participant. Subscale and total scores were collected for all 30 caregivers on the CQ and WeeFIM. Summary and subscale scores for the CHQ were only calculated for 25 of the 30 caregivers due to incomplete data. Four of these caregivers (participants 6, 10, 14, and 25) indicated specific questions were not applicable to themselves or their child. The fifth participant with incomplete CHQ data (participant 7) simply did not finish the last page of the assessment. This calculation for incomplete data sets is consistent with the procedure delineated by the CHQ User Manual (Landgraf et al. 1996). 604 Developmental Medicine & Child Neurology 2001, 43:

5 Results CQ CHQ CORRELATIONS Spearman s correlation coefficients between the CQ total score and the two CHQ summary scores ranged from r=0.217 to r=0.235 and were non-significant (Table II). There were significant relationships found between the CHQ subscale Parental Impact Time and the CQ total score (r=0.431) and CQ Physical subscale score (r=0.386). The CHQ subscale Family Cohesion and the CQ Interaction subscale (r=0.481) were also significantly related (see Table II). CHQ WEEFIM CORRELATIONS As anticipated by our original hypothesis, Spearman s correlation coefficients for the comparison of the total scores of the CHQ and WeeFIM were all non-significant. The correlation of the CHQ Physical summary score to the WeeFIM Selfcare and Mobility subscales were non-significant as was the CHQ Psychosocial summary score to the WeeFIM Cognition subscale score. CQ WEEFIM CORRELATIONS There was a significant relationship found between the CQ total score and the WeeFIM total score, r=0.460 (Table III). While this value is statistically significant, it suggests only a fair degree of correlation and would only account for 16% of the variance in scores (Portney and Watkins 1993). There were also significant relationships found between the various subscales of these two assessments ranging from r=0.388 to (see Table III). The CQ Comfort subscale was not included in the subscale analysis since there was no analogous WeeFIM subscale similar to the pain information in this CQ subscale. It should also be noted that the majority of participants did not report pain to be a significant problem for their child. Therefore, the impact of the comfort questions in the CQ is represented solely in the total score comparisons. Discussion When evaluating the efficacy of care, most research focuses on improvements in function rather than aspects of HRQL (Jette 1993). The results of this study provide information about the importance of measuring both HRQL and function in the clinic. Most of the children of caregivers in our study showed significant disability, yet quality of life issues did not correlate well with function. For example, the child of participant 29 required moderate-to-maximum levels of assistance to perform all self-care and mobility tasks, as measured by the WeeFIM. Nonetheless, the participant rated the child as very satisfied with his/her school abilities friendships and life overall as measured by the CHQ subscale focusing on selfesteem levels. Similar results were found in a related study of adults with spinal cord injury, which showed no correlation between functional level, as measured by the adult Functional Independence Measure, and life satisfaction (Fuhrer et al. 1992, Manns and Chad 1999). The CHQ Parental Impact Time subscale, designed to capture limitations in personal time experienced by the caregiver based on the child s health-related status (Landgraf et al. 1996), correlated significantly with the CQ total and Physical subscale. These correlations may help validate the CQ as a HQRL measure. For example, participant 26 found that the majority of tasks on the CQ were moderately difficult for her and her child to perform. On the CHQ this same caregiver indicated that her time was limited a lot because of the Table II: Total and subscale correlations examined for three assessments CQ Total CQ Physical CQ Interaction r r r CHQ Physical Summary CHQ Psychosocial Summary CHQ Parental Impact Time a a CHQ Parental Impact Emotional CHQ Family Activities CHQ Family Cohesion b a Correlation is significant at 0.05 (two-tailed). b Correlation is significant at 0.01 (two-tailed). Table III: Spearman s rho values for CQ and WeeFIM total and subscale scores CQ Total Personal Care Positioning/Transferring Interaction r r r r WeeFIM Total Self-care Mobility Cognition a a a b a Correlation is significant at 0.05 (two-tailed). b Correlation is significant at 0.01 (two-tailed). Health-Related Quality of Life and Functional Outcome Measures in CP Jane W Schneider et al. 605

6 child s physical health, emotional well-being, and behavior. For this participant, CQ scores accurately reflected the negative impact of the child s health status on the caregiver s time as measured by the standardized CHQ. Likewise, the CHQ- FC was developed to capture family relationships in general (Landgraf et al. 1996). The significant correlation of the CHQ-FC with the CQ Interaction subscale suggests the CQ Interaction scale may be measuring a similar aspect of a child s ability to get along with others. The CQ may reflect the impact of the child s physical condition on the child, the caregiver, or both. Vague CQ directions request that the caregiver rate how easy or difficult it is for you or your child to perform each task. It is unknown if the participants changed their perspective with every question, consistently answered from their own or the child s perspective, or answered from the perspective of both working collectively. This inconsistency could be eradicated by adding that each question should be rated from the viewpoint of whoever consistently does more of the task. The CHQ physical functioning questions might be less relevant to the quality of life in children with CP. Many parents indicated that the questions were too high level for their children and one parent stated that Our daughter is mentally handicapped and wheelchair bound, so many of these questions are not applicable. The questions asked about participation in activities such as playing soccer or bike riding. In fact, four of the five caregivers that did not complete the CHQ had children who were more severely involved motorically (GMFC levels IV V). The level of questions used in the CHQ, and probably in other generic HRQL measures, suggest that these assessments may be less clinically relevant for children with more severe neurological issues. In contrast, the CQ asks questions about physical functioning related to difficulty in performing activities of daily living such as dressing or performing transfers (see Appendix). Measures of HRQL in adult populations are most commonly related to activities of daily living (Finkelstein 1998). As the difficulty of performing daily tasks is also applicable to the quality of life of children with CP, the CQ may be more useful than the CHQ for this population (Finkelstein 1998). The CQ was originally devised for use with children after dorsal rhizotomy surgery when pain is a significant issue. The children of the caregivers in this study, however, had not undergone selective dorsal rhizotomy and most were followed regularly in the clinic for spasticity management. This may explain the high ratings in the Comfort section of the CQ, which suggests that pain was not a significant problem for their children. Nonetheless, as pain is a common finding in persons with CP (Schwartz et al. 1999) the Comfort section of the CQ is important to document the effect of pain on HRQL. The similarity of some of the questions in the CQ and the WeeFIM may explain the fair association found between the two instruments. The WeeFIM assesses the level of the child s independence in performance of daily tasks, while the CQ measures the amount of difficulty for the child and the caregiver when performing similar tasks. Levels of difficulty and of dependence might be expected to correlate only to a fair degree as was illustrated in this study. Also, the WeeFIM was not designed to measure community participation or psychosocial experience, which is included in the last section of the CQ. The best description of the CQ may be that it is a measure specific for children with CP and their caregivers that measures quality of life through questions related to activities of daily living. The CQ may be able to provide additional information that the WeeFIM does not. As an example, one participant (nr 11), whose child had severe CP (GMFC level V), consistently scored 1 of 7 on the WeeFIM Mobility scale indicating complete dependence of the child. The parent, however, indicated that specific items such as transferring in and out of a wheelchair and getting out of a car were more difficult than other related items in the Positioning/Transferring section of the CQ. Thus the CQ scales would help identify the most difficult tasks for the caregiver child unit, direct the goals of treatment, and quantify changes. The CQ could easily be integrated into the clinical setting since administration and scoring are relatively quick and easy. In fact, most participants in the study completed the CQ in less than 10 minutes. The CHQ is an established valid and reliable generic HRQL measure that is appropriate for the research setting and allows for comparisons across populations (Landgraf et al. 1996). However, the CHQ may be less applicable for clinical use in children with CP. Disease-specific issues (like positioning/transferring) were not addressed in the CHQ while other questions were not appropriate for many of the children with CP. Some caregivers chose to write not applicable for those items and thus summary scores could not be calculated. Without a summary score, a clinician would have a more difficult time understanding the results of the CHQ. Manually scoring each section was time consuming, and calculating a summary score required the use of a computer. Time for scoring was estimated to be 30 to 45 minutes for each child once the administrator was familiar with the scoring system. Additionally, completing the CHQ took approximately 15 to 20 minutes, which was longer than completing either the CQ or the WeeFIM. Consequently, this assessment may be less feasible for clinical use with this population than the CQ. In summary, because improving quality of life is one of the goals of intervention, clinicians and researchers alike should consider using some type of HRQL measure in addition to a functional assessment when evaluating children with CP. Information gained from HRQL measures, especially diseasespecific measures, can help focus clinicians on the areas that are most important for child/family functioning. Disease-specific measures such as the CQ include questions unique to a child s disability and thus more specifically evaluate the wellbeing of the child and the efficacy of intervention on quality of life (Dijkers 1999). Presently, disease-specific instruments are scarce and no other HRQL measures catering to children with CP have been found in the literature. The Caregiver Questionnaire shows potential as a quick and easy assessment of HRQL in children with CP, however, reliability and validity testing is needed before clinical use is advocated. Accepted for publication 8th March Acknowledgements We wish to thank Beverley Tann RN, Kathryn Danien PT, and Karen Paterson PT for their help in securing participants for the study. We also appreciate Jana Marogil and Janet Simpson for their efforts related to WeeFIM training. 606 Developmental Medicine & Child Neurology 2001, 43:

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Appendix: Caregiver questionnaire Patient s name: Name of person completing form: Date: Please rate how easy or difficult it is for you or your child to perform each of the following tasks by drawing a slash (/) across the line at the point that represents the difficulty you experience. If an item is not appropriate, circle NA. Personal Care 1. Putting on pants? Health-Related Quality of Life and Functional Outcome Measures in CP Jane W Schneider et al. 607

8 2. Taking off pants? 3. Changing diapers? 4. Cleaning child s bottom? 5. Ease of toileting? 6. Ease of bathing? 7. Ease of feeding? 8. How satisfied are you with your child s progress in the personal care area? Very Satisfied Not at all satisfied Positioning/Transferring 9. Ease of positioning in a wheelchair? 10. Ease of positioning out of a wheelchair? 11. Ease of transferring in and out of a wheelchair? 12. Ease of getting down to the floor? 13. Ease of getting up from the floor? 22. How important are these questions on health and comfort to your child s quality of life? Not Important Very Important Interaction/Communication 23. How easy is it for your child to play alone? 24. How easy is it for your child to play with other children? 25. How easy is it for your child to keep up with other children during play? 26. How easy is it for your child to be completely understood by those who know your child well? 27. How easy is it for your child to be completely understood by someone who is a stranger? 28. How satisfied are you with your child s abilities in the interaction/communication area? Very Satisfied Not at all satisfied 29. Describe your child Very Happy Very Unhappy The Caregiver Questionnaire was developed by Deborah Geabler-Spira MD, and adapted by Jane W Schneider PhD PT, Linda M Gurucharri MPT, and Allison L Gutierrez MPT. 14. Ease of putting on braces or positioning devices? 15. Ease of getting out of a car? 16. How satisfied are you with your child s progress in the positioning/transferring area? Very Satisfied Not at all satisfied Comfort 17. My child is usually healthy and active. Agree Disagree Does your child have pain? Circle one. Yes No If No, go to Question Is there pain or discomfort during position changes? Never Always 19. Is there pain or discomfort during diaper changes? Never Always NA 20. Does the pain or discomfort prevent your child from participating in school, various programs, or other activities? Never Always 21. Is your child using pain control medicine? Never Always 608 Developmental Medicine & Child Neurology 2001, 43: