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1 Swiss Paediatric Respiratory Research Group, Dept of Social and Preventive Medicine, University of Bern, Bern, Switzerland. 2 The Leicester Children's Asthma Centre, Division of Child Health, Dept of Infection, Immunity & Inflammation, University of Leicester, Leicester, UK.
CORRESPONDENCE: C. E. Kuehni, Swiss Paediatric Respiratory Research Group, Dept of Social and Preventive Medicine, University of Bern, Finkenhubelweg 11, CH-3012, Bern, Switzerland. Fax: 41 316313520. E-mail: kuehni{at}ispm.unibe.ch
Keywords: Childhood asthma, epidemiology, ethnic groups, questionnaires, social class, wheeze
Received: January 17, 2006
Accepted July 17, 2006
| ABSTRACT |
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In a questionnaire survey of a random population sample of 4,236 children aged 610 yrs, parents' definition of wheeze was assessed. Predictors of a correct definition were determined and the potential impact of incorrect answers on prevalence estimates from the survey was assessed.
Current wheeze was reported by 13.2% of children. Overall, 83.5% of parents correctly identified "whistling or squeaking" as the definition of wheeze; the proportion was higher for parents reporting wheezy children (90.4%). Frequent attacks of reported wheeze (adjusted odds ratio (OR) 3.0), maternal history of asthma (OR 1.5) and maternal education (OR 1.5) were significantly associated with a correct answer, while the converse was found for South Asian ethnicity (OR 0.6), first language not English (OR 0.6) and living in a deprived neighbourhood (OR 0.6).
In summary, the present study showed that misunderstanding could lead to an important bias in assessing the prevalence of wheeze, resulting in an underestimation in children from South Asian and deprived family backgrounds. Prevalence estimates for the most severe categories of wheeze might be less affected by this bias and questionnaire surveys on wheeze should incorporate measures of parents' understanding of the term wheeze.
Since the mid 1960s, the prevalence of childhood asthma has seen a high increase, levelled off and possibly begun to decline 14. These changes have defied convincing explanation and the possibility that some of the time trends could be artefactual cannot be excluded. Current wheeze is conventionally used as a proxy marker to determine asthma prevalence in population surveys, with asthma being by far the most common cause of childhood wheeze beyond the neonatal period 5. Alternative methods are unsatisfactory. Reported doctor's diagnosis is unreliable because diagnostic customs change with time 6 and objective measurements, such as lung function or bronchial responsiveness, are difficult to perform in large samples and results are not specific 7. Even clinicians rely primarily upon a history of wheeze provided by parents for diagnosing and managing asthma.
Population estimates of asthma are thus directly dependent on parents' understanding of the term "wheeze". Recent studies of hospital-based samples of children have challenged the validity of parent-reported wheeze, by showing that conceptual understanding of wheeze by parents differed from the definitions used by epidemiologists 8 and that parental understanding differed between ethnic groups 9. To avoid such misunderstandings, it has been suggested that the term wheeze should be explicitly defined in questionnaire surveys 8. Even so, it is not known whether parents adhere to the definition provided.
The present study utilised a recent questionnaire survey on respiratory symptoms in school children, which aimed to determine the scale, predictors and potential impact of parental misunderstanding of the term wheeze. Parents' knowledge of the term wheeze was investigated by assessing the description of the sound their child made when reported to have wheezed. The predictors of a correct definition were determined and the potential impact of incorrect answers on the results of the current survey was explored.
| METHODS |
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Questionnaire
At the top of the four-page questionnaire 1, 1315 a definition of wheeze was provided as "breathing that makes a high-pitched whistling or squeaking sound from the chest, not the throat". Current wheeze was then assessed with a validated question: "wheezing or whistling in the chest in the last 12 months?" 5. Parents reporting wheeze in their child were asked if the sound their child made when wheezing included the following: 1) squeaky or whistling noises; 2) wet, rattly noises; or 3) both whistling and rattly noises. For analysis, answers were categorised into two groups: those reporting whistling noises (with or without rattly noises) and those reporting only rattly noises.
At the end of the questionnaire, parents were asked if any of the following words meant the same as wheeze: rattly breathing; snoring; noises from the nose or throat during sleep; croup or stridor; whistling or squeaking noise in the chest; worrying dry cough; and moist or wet cough with phlegm. Every term could be answered separately as "yes", "no", or "don't know". For analysis, answers to these seven questions were summarised into three categories: 1) only whistling or squeaking noise accepted as a synonym for wheeze; 2) whistling or squeaking together with one or more other positive answer; and 3) whistling or squeaking not accepted as a synonym for wheeze.
The questionnaire also contained questions on severity and triggers of wheeze, environmental exposures, family history of atopy and socioeconomic conditions, measured at the individual level (duration of parental education) and area level (Townsend score, based on 1991 census data). Four families accepted an offer, written in Gujarati, Punjabi, Bengali and Urdu, of a phone interview in a South Asian language.
Analysis
There were <2% of answers missing for most questions on symptoms, and between 1.9 (whistling, squeaking noise) and 10.5% (croup/stridor) of answers missing in the questions on definition of wheeze. The answer to the question on noises heard when the child had been wheezing was missing in 2.8% of current wheezers and 27.1% of ever wheezers. For the analysis, children whose parents did not reply to the question on the definition of wheeze (121) were excluded, leaving 4,115 children for further analysis. Missing and "don't know" answers in questions on symptoms were coded "no" when sensitivity analyses showed that this did not influence the magnitude or direction of the findings, or else were entered in the models as separate answer categories.
To investigate which factors were associated with the current outcome of interest, multivariate logistic regression models were used, with results expressed as odds ratios (OR) with 95% confidence intervals (95% CI). All variables that were associated with prevalence of wheeze (p<0.05) in univariate analysis plus age, sex and ethnicity, were kept in the multivariate models.
| RESULTS |
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Description of sounds made by children reported to have wheezed
In total, 73.3% (688) of parents who reported that their child had ever wheezed said that their child made whistling noises when wheezing, and 26.7% (250) said their child made only rattly noises (table 2
). Among current wheezers (544), the proportions were 76.1 (414) and 23.9% (130) respectively. Predictors of an accurate description of the noises made when wheezing (i.e. whistling) were: male sex, age >8 yrs, sleep disturbance due to wheeze, a doctor's diagnosis of asthma in the child, maternal asthma and South Asian ethnicity. Neither deprivation nor fathers' education affected the accuracy of reported wheeze (table 2
).
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| DISCUSSION |
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Methodological issues
The strengths of the present study include the population-based sampling strategy, large sample size and inclusion of South Asian families, the largest ethnic minority group in the UK. In contrast to reports on hospital-based samples, the results can be used to interpret the accuracy of questionnaire surveys in unselected population samples. A limitation of the study is the relatively low response rate and the possibility that there might have been a learning effect, as the families had received three questionnaires within 5 yrs. Also, it is important to note that a definition of wheeze was included in the questionnaire. These factors are likely to have led to an overestimation of the correct understanding of the word wheeze in the surveyed Leicestershire families. As written questionnaires by their nature rely on both knowledge of the language and writing skills, the current results cannot be extrapolated to other survey methods such as video questionnaires.
Interpretation of findings
Of the parents reporting a wheezy child (13.2%), about a quarter did not describe having heard whistling or squeaking noises on the occasions their child had been wheezing. If it is assumed, very conservatively, that all of these are not true wheezers, the corrected prevalence estimate (scenario A) would be 10%. The proportion of such false positives did not vary much by ethnic or socioeconomic group.
Of the parents reporting no wheeze in their child (86.8%), a large proportion (17.5%, equalling 15.2% of the total population) did not know the correct definition of wheeze. Some of these might have given false-negative answers, either because the wheeze was inaudible or the whistling noise was actually heard by parents but not recognised as wheeze. As this group is relatively large, even a small proportion of false under-reporting of wheeze could have a great effect on the total population estimate of wheeze. Questionnaire surveys are, therefore, more likely to underestimate the true prevalence of wheeze, not the converse.
The present findings have particular implications for interpretation of differences in prevalence between subpopulations, as summarised in table 4
. With respect to ethnic groups, a recent systematic review concluded that the prevalence of wheeze and diagnosed asthma were lower in South Asian than white children in the UK, but that medical consultations and hospital admissions were more common 16. Differences in understanding of the term wheeze by respondents from different ethnic groups could help explain this discrepancy. In the present survey, the crude prevalence of current wheeze was similar in South Asian and white children (13.5 compared with 13.2%). This was also true for the proportion of children for whom there was both reporting of current wheeze and an accurate description of noises heard during wheezing (10.1 and 10%, respectively). However, 28.3% of South Asian parents compared with 11.1% of white parents did not know that wheeze meant whistling. Due to this large, potentially false-negative group, the true prevalence of wheeze in South Asian children, using the realistic scenario C could in fact be higher (14.2%) than in white children (11.4%). Similarly, the reported association between wheezing disease and deprivation in adults and schoolchildren 1720 may be partly misleading, since the accuracy with which wheeze was reported in the current study was lower in families living in deprived circumstances. Using scenario C, the proportion of current wheeze in the lowest (least deprived) and highest (most deprived) thirds of the Townsend score would be 10.8 and 16.1%, respectively, which increases the difference in prevalence between socioeconomic groups compared with the crude prevalence estimates (12.9 versus 15.8%). The most reassuring findings of the present survey were that understanding of the term wheeze and accuracy of symptom reporting rose with increasing frequency and severity of wheeze. Therefore, more stringent definitions of wheeze (severe wheeze) should be routinely used as additional outcome measures in epidemiological surveys in order to confirm their main findings.
Comparison with other studies
The present general population-based results confirm and supplement findings from hospital-based populations, which showed limited understanding of the term wheeze by parents 8, 9, 21. It has been suggested that a video questionnaire might avoid the problem of misunderstandings inherent to written questionnaires. The prevalence of wheeze as assessed by video questionnaires is lower than if assessed by written questionnaires, and the correlation between the two methods is not very good 22. However, video questionnaires were not better predictors of objective traits associated with asthma than written questionnaires 22.
Implications for future research and clinical practice
Future studies should interpret crude prevalence estimates of wheeze with more caution, especially when using the data to compare different ethnic groups and social classes. The main analyses, especially if the outcome of interest is current wheeze, should be confirmed by a secondary analysis including only those with more severe wheezing. The precision of prevalence and incidence estimates might be improved by inclusion of questions which determine parents' understanding of the term wheeze and descriptions of the sounds heard on the occasions when the child is reported to have been wheezing. Objective measurements (such as bronchial responsiveness or allergy tests) should be used to validate findings in subgroups of the population.
Another finding that might have implications for research and clinical practice is that significantly more parents of young females reported rattly noises in their child during a wheezing attack, compared to young males. This might imply differences in symptom presentation in young females and males, and could explain some of the apparent underdiagnosis and undertreatment of young females 10, 23, 24. For clinicians, who rely largely on parental reports of symptoms for managing wheezing children, it is essential to explore parental understanding of the term wheeze rather than accepting reports at face value.
In conclusion, the present study showed that the interpretation of questionnaire studies in childhood wheezing disorders is hampered by variation of parents' understanding and interpretation of the term wheeze. This uncertainty should be accounted for by both clinicians and researchers, especially when investigating sociodemographic and ethnic variations in asthma prevalence. Prevalence estimates for the most severe categories of wheeze might be less affected by this bias and questionnaire surveys on wheeze should incorporate measures of parents' understanding of the term wheeze.
| ACKNOWLEDGEMENTS |
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| REFERENCES |
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