Young patients with Crohn disease (CD) experience challenges arising from the symptoms, tests, and treatments of a chronic gastrointestinal disease. These are not just physical in nature; social and emotional issues can also affect quality of life (1–3).
Complications of CD may alter the appearance of young patients with CD, causing them to feel different from their peers. Treatments may also leave reminders of the disease, such as nasogastric tubes, ostomies, and scars from surgery (4). Weight loss may lead to patients being stigmatised as anorexic. Conversely, patients may experience excess weight gain resulting from prolonged steroid treatments. Patients with a gastrointestinal disease are subjected to invasions of privacy, and bowel symptoms may cause social embarrassment (3). Anxiety and depression have been shown to be associated with CD in both paediatric and adult patients (5). Because CD presents a range of challenges, many young patients will require psychological support (4).
Health-related quality of life (HRQoL) assessment represents how a patient perceives his or her physical, psychological, and social well-being, in the context of their culture and values (6). By taking into account multidimensional key aspects of an individual's life, it provides a more accurate measure of health than disease severity indexes (7). HRQoL scores may be used to determine resource allocation or assess the effectiveness of interventions (8). They may also bring to light any specific issues within certain domains. In paediatric patients with CD, it is vital to address any HRQoL issues because chronic illness can impair physical and psychosocial development (9) and lead to depression (10).
There are several paediatric quality of life scoring systems, some general and others that are disease specific. Previous investigators have ranked quality of life concerns of paediatric patients with inflammatory bowel disease (IBD) (8,11) to create disease-specific instruments for measuring HRQoL in children and adolescents. The subsequently developed IMPACT questionnaire had been validated as an inflammatory bowel disease–specific measure of HRQoL of paediatric patients in Canada (12), the United Kingdom (13), and the Netherlands (14). It was modified several times, and the IMPACT-III version has been used to assess HRQoL in the United States (15–19) and Australia (20).
CD has been shown to have a negative effect on the quality of life of affected paediatric patients in the United Kingdom (21,22). A New Zealand study (23) has qualitatively investigated CD from the perspective of 4 recently diagnosed adolescents. It illustrated the profound changes in the lives of young people after a diagnosis of CD; however, there is a lack of quantitative data on HRQoL in young patients with CD in New Zealand. With HRQoL being dependent on culture and societal values, it is not valid to extrapolate findings from other countries to New Zealand.
The purpose of the present study was to quantitatively determine the HRQoL status of paediatric patients with CD in the Wellington region using a previously validated questionnaire. In addition to providing a broad picture of the present situation, the study analyses patient and/or disease characteristics for correlation with HRQoL. An open-ended question allows patients to comment on issues of importance to them, providing qualitative information.
Patients in the Wellington region ages 9 to 18 years who had been diagnosed as having CD were included in the present cross-sectional study. A total of 18 subjects were identified, most from the Wellington Hospital laboratory database and 1 through contacting the Crohn and Colitis Support Group. Table 1 shows demographic and CD-related data for the participants (24).
The present study used the IMPACT-III questionnaire to measure HRQoL. This self-administered questionnaire includes 35 questions, spanning 6 domains of HRQoL (social functioning, bowel symptoms, emotional functioning, systemic symptoms, body image, and treatment/interventions). Each item has 5 possible responses on a Likert scale. The responses are summed to give a total HRQoL score, with a minimum possible value of 35 and a maximum possible value of 175. The questionnaire includes a final open-ended question asking the respondents to comment on anything else they believe is important or any other remarks.
The questionnaire was posted to patients ages 16 to 18 years, and to a caregiver of patients 9 to 15 years old. Informed written consent was gained from participants and from a caregiver of those in the younger category.
Because the IMPACT questionnaire had not previously been used in New Zealand, cross-cultural adaptation was required. This adaptation ensured that content validity of the questionnaire was maintained (25). Subjects were invited to meet with the researcher and participate in cognitive debriefing for this process. Debriefing followed the guidelines proposed by Beaton et al (25), with paraphrasing to determine whether each question and the response sets were understood and interpreted as intended.
Because of the small sample size and the possibility of outlier values, both the mean and median were calculated for the total HRQoL score and domain scores. Also for these reasons, nonparametric analysis methods were used. The Mann-Whitney U test was used to determine the relation between sex and total HRQoL scores; the Spearman rank correlation coefficient was used for the other patient characteristics. There was no attempt to compare domain scores because of differing representation.
To assess disease severity, the Paediatric Crohn Disease Activity Index, previously shown to be a sensitive and responsive measure of disease activity, was calculated from data collected at the patients’ most recent outpatient contact (26–28). The most recent outpatient contact was within 1 week of the questionnaire being filled in for all except 3 of the participants, with those completing in person having the questionnaire on the same day as the outpatient appointment. The remaining participants, who were not seen at the clinic within 1 week of the questionnaire, were seen at 21 days, 61 days, and 81 days before the clinic. All of these participants had quiescent disease and were not seen at the regular paediatric gastrointestinal clinic because their disease was stable. The New Zealand Deprivation Index, calculated from 9 census variables, was used to calculate a social deprivation score based on the residential address of the child (24). The present study received ethical approval from the central regional ethics committee.
The participation rate was 16 of 18 (89%). Half of the participants agreed to the cognitive debriefing; the remaining half completed the questionnaire and returned it by post. All 16 of the completed questionnaires were filled out correctly and included in the analysis. There was 1 missing value, which was addressed according to the IMPACT-III user's guide (28). Because the value was from one of the larger domains (bowel symptoms), it was entered as the average of that individual's scores for the other questions in that domain. Table 2 shows descriptive analysis of the results, with higher scores indicating a better quality of life.
The questions with the lowest mean scores were question 8 (effect of IBD on family; mean 2.4, median 2), question 22 (feelings about tests; mean 2.4, median 2.5), and question 5 (being bothered by having an illness that will not go away; mean 2.63, median 3). The questions with the highest mean scores were question 18 (whether it is harder to make friends because of IBD; mean 4.6, median 5), question 25 (afraid of not reaching the toilet in time in the last 2 weeks; mean 4.4, median 5), and question 23 (being bullied or left out of activities because of IBD; mean 4.3, median 5).
Mann-Whitney U test analysis showed no significant difference in the rank distribution of total HRQoL scores between boys (mean rank 8.56) and girls (mean rank 8.44, Mann-Whitney U 31.5, 2-tailed significance 0.958). Table 3 shows nonparametric analysis of correlation between patient characteristics and total HRQoL scores. Significant findings included a positive correlation between disease duration and quality of life (ρ = 0.534, sig.<0.05) and a negative correlation between disease activity and quality of life (ρ = −0.596, sig.<0.05).
Cognitive debriefing established that the vast majority of questions and all of the response sets caused no difficulty for participants; however, 3 participants expressed difficulty with question 19 (How often do you worry about your stool [bowel movement] containing blood?) from the bowel symptom domain. To understand the question, they either guessed the meaning of the word “stool” from the context of the question or asked its meaning. Cross-cultural adaptation of the questionnaire will take this into account.
Qualitative information from the open-ended question showed that the chronic nature of CD and its treatment caused the most distress. Some children mentioned that long treatment regimens were tiring, and that visual reminders of CD (eg, nasogastric tubes) cause embarrassment. Some of the patients did not know a person of a similar age who also had IBD, and their caregivers indicated a need for social support targeted toward children.
The present study is the first to produce quantitative data on HRQoL of paediatric patients with CD in New Zealand. Significant findings include a moderate to strong positive correlation between duration of CD and total HRQoL (ρ = 0.534, sig. <0.05). This may be the result of the relapsing and remitting nature of CD. Also, there was a moderate to strong negative correlation between the Paediatric Crohn Disease Activity Index and total HRQoL (ρ = −0.596, sig. <0.05), showing a relation between present disease activity and a lower quality of life. There was no correlation among HRQoL and age at diagnosis, patient age, or social deprivation score (New Zealand Deprivation Index (24))
These findings are in agreement with those of several previous studies on IBD in general. Otley et al indicated an improvement in HRQoL in the first year after diagnosis (29). The present study and another also found a negative effect of IBD severity on HRQoL scores.
Because all of the HRQoL assessments are subjective and use an arbitrary scale, the question arises as to whether the results of the present study indicated overall “good” or “bad” HRQoL scores. A comparison group was not possible because of the disease-specific nature of the IMPACT-III questionnaire; however, the goal of health care service delivery and treatment regimens should be to increase HRQoL from its baseline value. An effective way of doing this is to successfully control the disease activity itself (1), and the results of present study indicated that those with less active disease are more likely to have a better quality of life score.
The findings of the present study must be interpreted with caution because of the small sample size. We were only able to identify 18 patients between the ages of 9 and 18 years with CD. Wellington city has a population of 200,000 with adjacent urban areas increasing the wider Wellington area to 350,000. A survey undertaken between 2002 and 2003 by the New Zealand Paediatric Surveillance Unit showed that the incidence of paediatric CD was 1.6 of 100,000 for the Wellington region (30). Two patients did not participate, both for unknown reasons. It is possible that a good HRQoL would cause a patient to perceive participation as unnecessary, or that a bad HRQoL would cause too much distress to participate, leading to selection bias. Those who participated may have chosen a period of wellness in which to complete the questionnaire, which would overestimate HRQoL. The environment in which the questionnaire was completed was uncontrolled for half of the participants. For the other half, the researcher's presence may have unintentionally influenced some answers.
Despite limitations, there were clear areas of concern identified in the quantitative data and backed up by answers from the open-ended question. These were the effect of CD on family and having an illness that will not go away. Both of these findings indicate the scope for more psychosocial support to be given to children and their families, for example, through a child psychologist. Because the question about tests and treatments also received low scores, the present study indicates that more focus is required on minimising the psychosocial and emotional effects in this area. In the qualitative information, both parents and children indicated the need for more social support targeted toward children. Some support is given by the Wellington Crohn and Colitis Support Group, but this is not funded and dependent on the time and enthusiasm of volunteers. Some children with significant psychological distress are presently or have been engaged with the Child and Adolescent Mental Health Service, but there is a reasonably high threshold required to meet their criteria for ongoing support. Early preemptive psychosocial intervention may reduce the level of the child and family's distress and therefore the need for ongoing Child and Adolescent Mental Health Service involvement. Most of the patients have also received some support by paediatric nurses in the community. A paid position attending the psychosocial needs of young people would provide a more effective service and allow continuity of support.
Lynch and Spence (23) also suggested a need for more appropriate services to target psychosocial and emotional functioning of young patients with CD in New Zealand. One way of achieving this would be to promote advanced roles of nurses in this area. A US study (31) showed improvements in HRQoL after attending an IBD summer camp. The possibility of running a similar programme in New Zealand could be explored.
Because the IMPACT-III questionnaire will be adapted to a version that is validated for use in Australasia, larger studies and research including ulcerative colitis could use this questionnaire.
Among children ages 9 to 18 years with CD in the Wellington region, those with a longer duration of CD are more likely to have a higher HRQoL score, as measured by the IMPACT-III questionnaire. Those with more active disease are more likely to have a lower score. Patients and their caregivers indicated that the largest obstacles in coping with CD were its chronic nature and the treatments such as pills and nasogastric tubes. Some also indicated a need for age-specific social support. It is hoped that further studies will add to the knowledge of HRQoL of young patients with CD and use that knowledge to explore resource allocation and as a method of assessing efficacy of interventions.
The authors thank the Child Health Research Foundation for supporting this project. Also thanks to Dr Ross Wilson, Dr Kevin Pringle, Dr Tony Otley, Prof Peter Crampton, Dr James Stanley, the Crohn and Colitis Support Group of Wellington, and all of the patients and caregivers who participated in the present study.
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