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Measuring quality of life in the parents children with asthma

E. F. Juniper,* G. H. Guyatt, D. H. Feeny, I? J. Ferrie, L. E. Griffith and M. Townsend
Department of Clinical Epidemiology and Biostatistics, McMaster University Medical Centre, Hamilton, Ontario, Canada (E. F. Juniper, G. H. Guyatt, D. H. Feeny, P. J. Ferric, L. E. Griffith, M. Townsend); Department of Medicine, McMaster University Medical Centre, Hamilton, Ontario, Canada (G. H. Guyatt); Centre for Health Economics and Policy Analysis, McMaster University, Hamilton, Ontario, Canada (D. H. Feeny)

of

Parents and primary caregivers of children with asthma are limited in normal daily activities and experience anxieties and fears due to the childs illness. We have developed the Paediatric Asthma Caregivers Quality of Life Questionnaire (PACQLQ) to measure these impairments. The objective of this study was to evaluate the measurement properties of the PACQLQ. A Qweek single cohort study was conducted with assessments at 1, 5 and 9 weeks. Participants in the study were primary caregivers of 52 children (age 7-17 years) with symptomatic asthma, recruited from notices in the local media and paediatric asthma clinics. Caregivers completed the PACQLQ, Impact-on-Family Scale and Global Rating of Change Questionnaires. Patients completed the Paediatric Asthma Quality of Life Questionnaire and an asthma control questionnaire. Spirometry and P-agonist use were recorded. The PACQLQ was able to detect quality of life changes in those caregivers who changed (p < 0.001) and to differentiate these from the caregivers whose quality of life remained stable (p < 0.0001). The PACQLQ is reproducible in subjects who are stable (ICC=O.84), and showed acceptable levels of longitudinal and cross-sectional correlations with the childs asthma status and health-related quality of life and with other measures of caregiver healthrelated quality of life. The PACQLQ functions well as both an evaluative and a discriminative instrument. Key words: Asthma; paediatrics health-related quality of life;

Introduction
Interest in the impact of illness on day-to-day function is leading investigators to include both diseasespecific and generic health-related quality of life (HRQOL) questionnaires in a broad range of clinical studies. However, illness not only has an impact on patients but also affects the quality of life of people associated with the patient. In childhood illnesses the family, and particularly the primary caregiver, may face a considerable burden. In a recent study, we showed that the primary caregivers of children with asthma are limited in their own normal daily activities and also experience anxieties and emotional stresses as a result of their childrens asthma. On the basis of these results, we developed the Paediatric Asthma Caregivers Quality of Life Questionnaire using methodsz3 that we have applied in the development of over 20 diseasespecific HRQOL questionnaires. In this paper, we briefly describe the methods used for the development of the questionnaire and present in detail the methods and results of a study in which we examined the measurement properties of the Paediatric Asthma Caregivers Quality of Life Questionnaire. We addressed four questions: (1) In caregivers whose HRQOL remains stable, what is the reliability of the questionnaire? (2) In caregivers whose HRQOL changes, are we able to detect these changes even if the changes are small (responsiveness)? (3) What change in score can be considered important? (4) Is the questionnaire valid, i.e. does it actually measure the HRQOL of the caregivers of children with asthma?

To whom correspondence should be addressed at Department of Clinical Epidemiology and Biostatistics, McMaster University Medical Centre, 1200 Main Street West, Hamilton, Ontario, Canada, L8N 325. Phone: 905-525-9140 x 22153; Fax: 905-577-0017; email: Juniper@fhs.mcmaster.ca
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E. E @@er et al.

Methods
Participants
We have described the patient population who participated in this study and its basic methods in another publication! In brief, we recruited 52 children with current symptoms of asthma and their primary caregiver from local paediatric asthma clinics, notices in the local media and through patients who had participated in our adult asthma research studies. The children were between 7 and 17 years and represented a wide range of asthma severity as judged by medication requirements. At the time of enrollment, all children were symptomatic and had no other illness that had an impact on quality of life. We recruited the primary caregiver, usually a parent, who lived with the child at least 75% of the time. Children over 12 years of age and all the caregivers signed an informed consent that had been approved by the McMaster University Faculty of Health Sciences Ethics Committee.

Study design
Patients and their caregivers attended the clinic at enrollment and after 1, 5 and 9 weeks. At each visit, the caregiver completed the Paediatric Asthma Caregivers Quality of Life Questionnaire and the Impact-on-Family Scale. At each follow-up visit, the caregiver completed three global rating of change questionnaires relating to changes in both the childs asthma and their own quality of life since their previous visit. At each clinic visit, the child completed the Paediatric Asthma Quality of Life Questionnaire,4 a clinical asthma control questionnaire6 and had spirometry measured before and after a bronchodilator. For one week before each of the follow-up visits, the children made measurements of peak expiratory flow each morning and completed an asthma symptom and medication diary each morning and evening.

caregivers of children with asthma; (2) include both physical and emotional impairments; (3) be reproducible when the caregivers quality of life is in a stable state; (4) be responsive to change in the caregivers quality of life even if that change is small; (5) be valid, i.e. actually measure quality of life in the caregivers of children with asthma. We have described, in another publication, the process by which we determined the problems experienced by parents of asthmatic children. In brief, a pool of 69 items was generated from unstructured interviews with parents of children with asthma, a literature review and discussion with health professionals. We asked 100 primary caregivers to identify which of the 69 items they had experienced in the last year. For each positively identified item, the caregiver rated the importance on a 5-point scale (l=does not bother me much, 5=bothers me very, very much). In general, items identified most frequently and rated most bothersome were those included in the questionnaire. Responses to each item in the Paediatric Asthma Caregivers Quality of Life Questionnaire are given on a 7-point scale where 1 represents severe impairment and 7 represents no impairment. We weight individual items within the questionnaire equally and express the results as the mean score per item for each of the domains (emotional function and activity limitation) as well as for overall quality of life. Therefore, both the domain and overah scores range from l-7. The questionnaire asks caregivers to recall impairments experienced during the previous week taking into account, at follow-up visits, their response at the prior visit. We pre-tested the Paediatric Asthma Caregivers Quality of Life Questionnaire in a group of 10 caregivers to ensure ease of completion and comprehension. Paediatric Asthma Quality of Life Questionnaire4. We have recently developed and tested this questionnaire. It includes 23 items in three domains (symptoms, activity limitation and emotional function) and measures HRQOL in patients with asthma age 7-17 years. We have demonstrated that in patients whose HRQOL is stable, it is reliable (intraclass correlation coefficient of 0.84). It is also responsive in that it can detect changes in patients whose HRQOL has changed and is able to differentiate between stable and unstable patients. Correlations between the instrument and both conventional clinical asthma measures and generic HRQOL measures were close to predicted for both longitudinal (related to use as an evaluative instrument) and cross-sectional (related to use as a discriminative instrument) validation.

Quality

of life outcome

measures

Paediatric Asthma Caregivers Quality of Life Questionnaire. The Paediatric Asthma Caregivers Quality of Life Questionnaire is a self-administered 13 item instrument. Four items concern activity limitations and nine concern emotional function. We designed the instrument to meet the following criteria: (1) measure areas of function that are important to the primary

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HRQOL in asthma parents Impact-on-Family Scal$. This is a 24item quality of life instrument that evaluates the impact that a childs illness has on family function. Initially, a pool of 190 potential items was generated through discussion with New York City parents and this was reduced to 24 items by an expert panel and factor analysis. The final questionnaire has four domains, financial (n = 4), family/social (n = 9), personal strain (n = 6), mastery (n = 5). Responses to each item are given on a 4point Likert scale (strongly agree to strongly disagree). Internal consistency (Cronbachs 01)for overall impact and for each domain ranges from 0.60-0.88. Global Rating of Change Questionnaires. These questionnaires ask about changes in quality of life since the previous clinic visit.* At each follow-up visit, we asked caregivers whether they had experienced any change in their overall quality of life, emotions or activity limitations related to their childrens asthma since their previous clinic visit. Responses are scored on a 15-point scale from -7 (a very great deal worse) to 0 (no change) to +7 (a very great deal better). (Maximum score = 6.)

Caregivers

perception

of childs asthma

At each follow-up clinic visit, we asked caregivers whether there had been any change in their childs asthma symptoms since the previous visit. They responded on a 1Spoint scale from -7 (a very great deal worse) to 0 (no change) to +7 (a very great deal better).

Classifying caregivers or unchanged

as changed

Asthma

clinical

outcome

measures

There were two study periods (2-5 weeks and 6-9 weeks). For each period, we categorized caregivers either as having stayed the same (Group A) or changed (Group B). If caregivers scored -1, 0 or +l on the global rating of change questionnaires (overall quality of life, activity limitations and emotional function), they were considered to have stayed the same and if they scored between -7 and -2 or between +2 and +7 they were considered to have changed.

For one week before each clinic visit, on rising each morning and before taking any asthma medication, the children made three peak expiratory flow rate measurements and recorded the highest value. They recorded whether they had been woken during the night by their asthma, whether they had asthma symptoms when they woke in the morning and how many puffs of inhaled P-agonist they used during the night. At bedtime, they recorded whether they had experienced any limitation in their normal daily activities as a result of their asthma, whether they had produced any sputum and how many puffs of inhaled B-agonist they had used since getting up in the morning. Patients reframed from bronchodilator use for at least 6 hours before each clinic visit. We measured spirometry before and 20 minutes after two puffs of salbutamol (200 pg). At each clinic visit, an asthma control questionnaire was completed.6 This is a composite of asthma symptoms and B-agonist use during the past week (diary) and clinic spirometry. One point is scored for each of the following present on one or more days: (1) awoken by symptoms at night; (2) awoke with symptoms in the morning; (3) sputum; (4) limitation of activities and (5) P-agonist use more than four times per day One point was scored if clinic FEVl prebronchodilator was less than 70% predicted.

Testing the measurement

properties

General approach. Health-related quality of life measurement instruments may have one or more purposes. An evaluative instrument is designed to measure the magnitude of longitudinal change in an individual or group. Evaluative instruments must be responsive (able to detect important within-subject changes, even if they are small) and demonstrate longitudinal construct validity (appropriate correlations between changes in the new questionnaire and changes in other measures). A discriminative instrument is designed to distinguish between people at a single point in time. Discriminative instruments require high reliability (high ratio of differences between subjects to difference within subjects) and cross-sectional construct validity (appropriate correlations between established rating scales and the instrument being tested). We tested both the evaluative and discriminative properties of the Paediatric Asthma Caregivers Quality of Life Questionnaire. Evaluative properties. We examined the responsiveness of the Paediatric Asthma Caregivers Quality of Life Questionnaire in three ways. First, we assessed the ability of the instrument to detect within-subject

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E. E Juniper et al. changes in subjects who changed (Group B) using a paired t-test. Second, we examined the ability of the instrument to distinguish between subjects who remained stable (Group A) and those who changed (Group B) using an unpaired t-test of the differences between the beginning and end of each period. For both of these estimations, symmetry of the data from those who had improved and deteriorated justified our changing the sign in those who deteriorated. Third, we calculated a responsiveness index from the minimal important difference and the pooled withinsubject standard deviation from both Groups A and B. The minimal important difference is the mean difference in score in those who score -3, -2, +2 or +3 on the global rating of change.* From the responsiveness index, we calculated sample sizes for parallel group and cross-over study designs for various levels of o! and p error rates. We assessed longitudinal construct validity by correlating within-subject changes in the caregivers quality of life scores over a four week period with within-subject changes in the childs health-related quality of life, indices of the childs clinical asthma severity and the caregivers generic quality of life. We also examined the correlation of changes in quality of life scores with the global ratings of change. Before analyzing the data, three clinicians made a priori predictions of where we should expect to find correlations if we were truly measuring quality of life in these caregivers. Discriminarive properties. We used data from caregivers who were stable between consecutive clinic visits (Group A) to determine the reliability of the instrument. If a caregiver was stable throughout the entire study (periods 1 and 2), one set of data was randomly selected for analysis. We express the variance over 4 weeks as the within-subject standard deviation of change. The ratio of the between-subject variance and the total variance has been expressed as an intraclass correlation coefficient. We examined cross-sectional construct validity by correlating caregiver quality of lifescores at each clinic visit with the measures of the childs asthma severity and with generic caregiver quality of life scores. Once again, three clinicians made predictions before the analysis.

Results
All fifty-two children and their caregivers finished the study and provided complete data sets. Fortyseven (90.4%) of the caregivers were female and five were male (9.6%) and their ages ranged from 30-63 years (mean 40.94, sd 5.6 years). The questionnaire took between 3 and 5 minutes to complete.

Evaluative

properties

Twenty-three caregivers contributed 31 sets of observations to the changed category (Group 8) for overall quality of life. Twenty-six caregivers contributed 36 observations to the emotional function domains and 17 contributed 22 observations to the activity limitation domain. The mean change in caregiver overall quality of life over a four week period was 0.72 (sd = 0.69) with similar changes in both the emotional function and activity limitation domains (Table 1). Not only were these within-subject changes significant (p < 0.0003) but the changes in Group B were also significantly different from the changes in score in the caregivers who reported staying stable (Group A) (p < 0.0001). The minimal important difference for overall caregiver quality of life was 0.50 with similar values for the emotional function domain (0.64) and the activity limitation domain (0.67) (Table 2). For moderate changes in quality of life (global rating = 4 and 5), the mean change in overall quality of life score was 1.12 with similar scores in the two domains. For the

Table 1. Responsiveness: The ability of the questionnaire to detect change Change in caregiver quality of life score** Stable subjects Subjects who changed Difference (A vs. 6) (Group A) (Group 6) p value -0.03 -0.02 -0.01 0.72* 0.71. 1.80* <0.0001 co.ooo1 0.0003

Domain Overall QOL Emotional function Activity limitation

*Within-subject change in score : p c 0.001 **Scores are expressed as the change in mean score per item

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Table 2. Change in caregivers quality of life scores vs. global rating of change -1, 0 and 1 Change in quality of life score Overall QOL Emotional function Activity limitation -0.03 n=72 -0.02 n=67 -0.01 n=80 Global rating of change -3, -2, 2 and 3 -5, -4, 4 and 6 0.50 n=l6 0.64 n=l8 0.67 n=9 1.12 n=12 0.92 n=14 1.05 n=ll -7, -6, 6 and 7 0.36 n=3 0.26 n=4 0.00 n=2

Table 3. Sample sizes for clinical trials Type 1 and type 2 error rates Study design Parallel group: n per group Overall Emotions Activities Cross-over: n of pairs Overall Emotions Activities a = 0.05 (one-sided) p = 0.1 p = 0.05 21 16 18 11 9 10 26 20 22 14 11 12 a = 0.05 (two-sided) p = 0.1 p = 0.05 25 19 22 13 10 12 31 23 26 16 12 14

Based on: Overall: minimal important difference = 0.50; sd = 0.51 Emotions: minimal important difference = 0.64; sd = 0.56 Activities: minimal important difference = 0.67; sd = 0.63

Table 4. Longitudinal construct validity-f (all subjects) Change in caregiver Emotional function Change in caregiver burden of illness Overall Family/social Personal strain Mastery Change in childs asthma seventy FEVI % predicted (pre-bd) Peak Expiratory Flow Rate (am) Clinical asthma control P-agonist use Caregiver global ratings of change Emotions Activities Caregiver perception of change in childs asthma survey A priori predictions:
l

quality of life Activity limitation -0.36 -0.38 -0.31 0.02 0.12 0.33* -0.23* -0.11 0.50 0.44*** 0.41
l

-0.39 -0.35 -0.24 -0.0007 0.03 0.31 -0.10 -0.22

0.55*** 0.46 0.52

r = 0.20-0.35; ** r = 0.35-0.50; *** r > 0.50 (no star indicates correlations about which we made no a priori predictions).

t Pearson correlation coefficient

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E. E Juniper et al. large changes (global rating=6 and 7), we encountered inconsistent changes in score (n=3). The responsiveness index for overall quality of life was 0.98 (emotional function=1.14 and activity limitation =1.07). From these values, we have calculated the sample sizes required for different a and p error rates in both parallel group and cross-over study designs (Table 3). Table 4 shows the observed and predicted correlations between changes in the domains of the Paediatric Asthma Caregivers Quality of Life Questionnaire and changes in the generic caregiver burden questionnaire and changes in the childs clinical asthma status. participant contributed to the estimation of reliability, the within-subject standard deviation of change was 0.31 for overall quality of life and the intraclass correlation coefficient was 0.85. Similar values were observed for the two domains (Table 5). The observed and predicted cross-sectional correlations supporting the discriminative validity of the questionnaire are shown in Table 6.

Discussion
The results of this study suggest that the Paediatric Asthma Caregivers Quality of Life Questionnaire performs well both as an evaluative instrument for clinical trials and as a discriminative instrument for cross-sectional surveys. The questionnaire proved highly responsive to within-subject changes over tune with the result that small sample sizes are required to detect changes that are small but considered important by caregivers. The instrument is also very reproducible with a low within-subject variance that results in a high degree of reliability in being able to detect differences in quality of life between caregivers

Discriminative

Properties

Forty-four caregivers contributed 72 sets of observations to the stable category (Group A) for overall quality of life. Forty-two caregivers contributed 67 observations to the emotional function domains and 46 contributed 80 observations to the activity limitation domain. When one set of observations per

Table 5. Reliability of quality of life scores in subjects with stable asthma (Group A) Domain Overall QOL Emotional function Activity limitation Within-subject standard deviation 0.31 0.36 0.39 Between-subject standard deviation 0.75 0.71 0.88 lntraclass correlation coefficient 0.85 0.80 0.84

Table 6. Cross-sectional construct validityt Caregiver quality of life Emotional function Activity limitation Caregiver burden of illness Overall Family/social Personal strain Mastery Childs asthma severity FEVI % predicted (pre-bd) Peak Expiratory Flow Rate (am) Clinical asthma control P-agonist use A @on predictions:
l l

-0.54 -0.56 -0.56* 0.28* 0.25 0.31 -0.29* -0.22*

-0.65 -0.62 -0.70 0.26 0.26 0.37 -0.30. -0.28

r = 0.20-0.35; ** r = 0.35450; ** r > 0.50 (no star indicates correlations about which we made no a priori predictions).

t Pearson correlation coefficient

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with different degrees of quality of life impairment. The high reliability and responsiveness of the instrument confirms we are measuring something, but is it quality of life related to childrens asthma? With respect to its evaluative validity, we observed the predicted moderate to high correlation with global ratings of change, and the same moderate to high correlation (this time higher than we predicted) with parental rating of change in the childrens severity of asthma. We observed the predicted weak correlations between change in questionnaire score and change in peak flow rates and asthma control but changes in questionnaire score with changes in forced expired volume in one second (FEVI) were lower than predicted. We interpret these results as being consistent with the questionnaire actually measuring changes in caregiver quality of life but reflecting the inadequacy of change in FEVl as a measure of change in asthma severity. With respect to its function as a discriminative instrument correlations between the Paediatric Asthma Caregivers Quality of Life Questionnaire and the Impact on Family Scale either met or were substantially higher than we predicted. We interpret these results as suggesting that the specific problems associated with caring for a child with asthma are more closely related to general problems of looking after a sick child than we anticipated. Correlations between asthma severity and questionnaire score were either in the predicted low range or somewhat lower than predicted. Once again, these results provide moderate support for the validity of the questionnaire in distinguishing between parents with more and less impairment quality of life as a result of their childs asthma. The limitations of our study include the relatively small sample size and our recruitment of a the population from a group of parents familiar with the research process. Testing the questionnaire in other settings, with further examination of its validity, would increase our confidence in its generalizability. Nevertheless, the strong reliability and responsiveness of our questionnaire, and the supportive evidence of its validity, suggest that investigators interested in measuring the impact of childhood asthma on caregivers can gain important insights through use of the new questioMaire.

References
1. Townsend M, Feeny DH, Guyatt GH, Furlong WJ, Seip AE, Dolovich J. Evaluation of the burden of illness for pediatric asthmatic patients and their parents. Annals Afkgy 1991; 67: 403-408. 2. Kirshner B, Guyatt GH. A methodological framework for assessing health indices. J Chron Dis 1985; 38: 27-36. 3. Juniper EF, Guyatt GH, Jaeschke R. How to develop and validate a new quality of life instrument. In: Spilker B, ed. Quality of Life and Pharmacoeconomics in Clinical Trials. Second Edition. New York: Raven Press Ltd, 1995: 49-56. 4. Juniper EF, Guyatt GH, Feeny DH, Ferrie PJ, Griffith LE, Townsend M. Measuring quality of life in children with asthma. Qua1 Life Res 1996: 5: 35-46. 5. Stem REK, Riessman CK. The development of an impacton-family scale: Preliminary findings. Med Care 1980; 18: 465-472. 6. Juniper EF, Kline PA, Vanzeileghem MA, Ramsdale EH, OByme PM, Hargreave FE. Long-term effect of inhaled corticosteroid (budesonide) on airway hyperresponsiveness and clinical asthma severity in nonsteroid-dependent asthmatics. Am Rev Respir Dis 1990; 142: 832-836. 7. Guyatt GH, Townsend M, Keller JL, Singer J. Should study subjects see their previous responses? Data from a randomised control trial. j C/in Epidemiol 1989; 42: 913-920. 8. Juniper EF, Guyatt GH, Willan A, Griffith LE. Determining a minimal important change in a disease-specific quality of life instrument. J Clin Epidemiol 1994; 47: 81-87. 9. Guyatt GH, Feeny DH, Patrick DL. Measuring healthrelated quality of life: Basic Science Review. Annals Int Med 1993; 70: 225430. 10. Guyatt GH, Walter S, Norman G. Measuring change over time: assessing the usefulness of evaluative instruments. J Chron Dis 1987; 40: 171-178. 11. Guyatt GH, Kirshner B, Jaeschke R. Measuring health

status: What are the necessarymeasurement properties.

] Clin Epidemioll992; 45: 1341-1345. 12. Malo JL, LArcheveque J, Trudeau C, dAquino C, Cartier A. Should we monitor peak expiratory flow rates or record symptoms with a simple diary in the management of asthma? 1 Allergy Chin Immunol 1993; 91: 702-709.

(Received 23 August 1995; accepted 25 September 1995)

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Paediatric

Asthma

Caregivers

Quality of Life Questionnaire

This questionnaire is designed to find out how you have been during the last week. We want to know about the ways in which your childs asthma has interfered with your normal daily activities and how this has made you feel. Please answer each question by placing an x in the appropriate box. You may only mark one box per question. 1. 2. 3. 4. 5. 6. 7. 8. 9. 10. 11. 12. 13. During the past week, how often did you feel helpless or frightened cough, wheeze, or breathlessness? when your child experienced

During the past week, how often did your family need to change plans because of your childs asthma? During the past week, how often did you feel frustrated due to asthma? or impatient because your child was irritable

During the past week, how often did your childs asthma interfere with your job or work around the house? During the past week, how often did you feel upset because of your childs cough, wheeze, or breathlessness? During the past week, how often did you have sleepless nights because of your childs asthma? During the past week, how often were you bothered because your childs asthma interfered family relationships? with

During the past week, how often were you awakened during the night because of your childs asthma? During the past week, how often did you feel angry that your child has asthma? During the past week, how worried daily activities? or concerned were you about your childs performance of normal

During the past week, how worried or concerned were you about your childs asthma medications and side effects? During the past week, how worried or concerned were you about being overprotective of your child?

During the past week, how worried or concerned were you about your child being able to lead a normal life?

Response Items Z-9 1. 2. 3. 4. 5. 6. 7.

Options Items IO-13 1. Very, Very Worried or concerned 2. Very worried or concerned 3. Fairly worried or concerned 4. Somewhat worried or concerned 5. A little worried or concerned 6. Hardly worried or concerned 7. Not worried or concerned

All of the time Most of the time Quit often Some of the time Once in a while Hardly any of the time None of the time

Domains Emotional Activities Function Items 1, 3, 5, 7, 9, 10-13. Items 2, 4, 6, 8

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