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The quality of life of home-ventilated children and their primary caregivers plus the associated social and economic burdens: a prospective study
  1. Michael Seear,
  2. Akshat Kapur,
  3. David Wensley,
  4. Kelly Morrison,
  5. Ariana Behroozi
  1. Division of Respiratory Medicine, BC's Children's Hospital, Vancouver, Canada
  1. Correspondence to Dr Michael Seear, Division of Respiratory Medicine, BC'S Children's Hospital, 4480 Oak Street, Vancouver, British Columbia, Canada V6H 3V4; mseear{at}cw.bc.ca

Abstract

Background Despite the obvious challenges faced by families caring for children on home ventilation, there is surprisingly little research into the details of their daily lives. In particular, little is known about the quality of life of the child and caregiver plus the associated social and economic burdens of care.

Methods We prospectively studied 90 families enrolled in a paediatric home ventilation service in British Columbia. In the clinic, we recorded demographic information, patient acuity score and quality of life for patient and caregiver using standardised questionnaires. Parents then monitored social and financial costs of care at home over the subsequent 8 weeks. These data were collected by telephone at 1 and 2 months.

Results Most children led rich active lives. Camping trips, wheelchair sports and foreign travel were the norm, not the exception. Over 90% assessed the burden of care as mild or moderate. Government support covers medical expenses and home nursing (median 32 h/week, IQR 0–62.5 h). Monthly unreimbursed family expenses were low (median $87.7, IQR $15.3–$472). Despite this, nearly 25% of primary caregivers assessed burden of care as severe and over 50% had chronic illnesses requiring daily medication (principally depression, anxiety and arthritis). Quality of life for children or caregivers did not correlate with income or education.

Interpretation Home ventilation of complex children is a successful strategy but it places significant strain on the primary caregiver. Specific attention to the physical and mental health of the caregiver should be an integral part of the management of home-ventilated children.

Trial registration number NCT01863992.

  • Multidisciplinary team-care
  • Patient perspective
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What is already known on this subject

  • Medical advances have resulted in growing numbers of technology-dependent children, roughly 5% of whom require ventilatory support at home. Although these children consume medical resources out of proportion to their numbers, there is surprisingly little research available to guide management decisions.

  • In particular, little is known about the quality of life of the affected children and their primary caregivers or the daily social and economic burdens these families face.

What this study adds

  • Home ventilation is a successful medical strategy. Over 90% of children assess the adverse effects of home care as mild or moderate and over 80% of school-age children attend regular school. However, this is achieved at significant expense of the caregiver's health.

  • One in four of the primary caregivers assessed the adverse effects of daily care to be severe and over 50% had significant chronic illnesses.

  • The health of the primary caregiver is essential for the health of the ventilated child. Medical care directed at the caregiver should be an integral part of the model of care for families caring for a child on home ventilation.

Introduction

Extreme disability is not synonymous with unhappiness….Dr Robert Zachary.1

Over the last 30 years, there have been three separate developments that, taken together, have had a profound effect on the care of children with major medical challenges. The first, typified by Dr Zachary's quote above, was a fundamental change in attitude towards children with severe disabilities.2 He was specifically writing about spina bifida but the full range of conditions affected by this revolution in care is enormous.3 Subsequent concerns about hospital costs and neurodevelopmental delay among institutionalised children led to the second development, which was to shift care to the child's home.4 Lastly, was the development of portable medical devices (particularly non-invasive ventilators), plus modern batteries, that allowed technology-dependent children to live an active mobile life.5 The final result has been steadily growing numbers of children with complex health conditions who are cared for at home, usually by a parent.6

Although definitions vary, recent database studies from the USA and Canada estimate that between 0.4%7 and 0.67%8 of North American children now have technology-dependent chronic health conditions. Although they constitute <1% of the paediatric population in Ontario, this group consumes nearly one-third of the Province's total child health expenditure.8 Paediatric home ventilation clinics have developed to meet this growing demand,9 ,10 but, even in a prosperous country like Canada, services for children with complex health problems remain fragmented and unstandardised across the country.6 ,11 Based on our own study,12 the prevalence of children requiring home ventilatory support in British Columbia is 1–2 per 10 000 children or roughly 5% of the total group with complex home care needs. In British Columbia, there has been a centralised home ventilation service for the Province's roughly 900 000 children for over 25 years.12 Enrolled families receive generous government support that covers home nursing, equipment purchase, medications and regular supplies.

While it is obvious that caring for a child with medical problems is a serious challenge for any family,13 there is surprisingly little quantitative research aimed either at children who need home ventilation14 or their caregivers and families.15 In order to improve our own clinical practice, we wanted to understand the impact of home ventilation on the daily lives of families enrolled in our clinic. Using validated questionnaires, we examined the adverse effects of home ventilation on the affected child and the primary caregiver. We also studied the health-related social and economic costs carried by the family. In addition, we took this opportunity to develop a preliminary paediatric home ventilation workload score suitable for standardising research in this patient population.

Methods

Patient selection

After permission was provided by the research ethics committee of the University of British Columbia, children and their primary caregivers were prospectively enrolled from the home ventilation clinic at BC's Children's Hospital. Written informed consent allowing us to collect questionnaire data was obtained from all children over 16 years of age. For all others, informed consent was provided by a legal guardian. Where possible, informed assent was obtained from all children below 16 years using age-appropriate forms. All children in British Columbia who were using regular invasive or non-invasive ventilatory support at home were eligible for study; there were no exclusion criteria other than refusal to join the study. We did not calculate the sample size. During the 9-month study period, we enrolled as many patients as possible so that the final study sample (table 1) would be representative of the very wide variations in diagnosis and acuity among our total of nearly 140 patients.

Table 1

The principal clinical characteristics of the children enrolled in the study

Study design

This was a prospective, questionnaire-based observational study. It consisted of an interview in the clinic, followed by two telephone calls at home. The initial questionnaires covered basic epidemiological data and patient workload plus the quality of life for the child and primary caregiver. Families were then shown how to use daily recording sheets which they filled in at home. These collected regular information on the social and economic impacts of home ventilation. The recorded data were collected by telephone calls at 1 and 2 months after the first visit. All study information was collected directly from the families. No information was obtained from postal questionnaires or patient databases. The following questionnaires were used:

  • Demographic information and patient workload score.

  • In addition to collecting basic demographic data for the affected child and primary caregiver, we also calculated a score representing our assessment of the caregiver's workload. While some jurisdictions use workload scores to guide reimbursement and suitability for home nursing,16 there is no validated acuity score suitable for research into technology-dependent children. We developed a preliminary acuity measure based on attributing scores for six elements of the child's daily care (table 2). The predictive value of the workload score was tested by comparing values against the caregiver's record of hours devoted to patient care each month.

  • Paediatric quality of life.

  • We assessed the impact of home ventilation on children with the widely used Paediatric Quality of Life Inventory (PedsQL 4.0).17 The PedsQL covers the widest range of patients by having eight separate forms designed for different age ranges and abilities. There are three self-reported forms (covering ages 5–7 years, 8–12 years, 13–18 years) and five caregiver proxy forms for children unable to answer questions (covering ages from infancy to 18 years). Questions assess the impact of the child's disease on daily living. Answers range from 0 (never a problem) to 4 (nearly always a problem). Scores for the different age range questionnaires are standardised by expressing them as a percentage of the total. The final score runs from 0% to 100%. Increasing values represent greater adverse impact of home ventilation and lower quality of life. PedsQL has been used to measure the adverse effects of a wide range of paediatric conditions, including children with severe neuromuscular diseases cared for at home.18

  • Caregiver quality of life.

  • The impact of home ventilation on the main caregiver was assessed using the validated Caregiver Impact Scale (CIS).19 It has been used to assess the effects of home care on the primary caregiver across a wide range of conditions, including home ventilation.20 The questionnaire examines how caregiving responsibilities interfere with 13 aspects of daily living. Each is scored from 0 (not at all) to 6 (very much). In order to remain consistent with PedsQL, the final score, in points, is expressed as a percentage of the maximum. As with the PedsQL score, increasing values indicate worsening adverse effects of caregiving.

  • Social and economic costs of home ventilation.

  • Information on the social and economic impacts of home ventilation was collected prospectively using the validated Ambulatory and Home Care Record (AHCR).21 It has been used to calculate the costs of home care for a range of medical conditions.22 The AHCR records unreimbursed health-related costs such as medications, travel to appointments, paid care providers and consumable medical supplies. It also tracks the caregiver's total time commitment. The recorded data were collected by telephone calls at 1 and 2 months after the initial interview. All costs were expressed in Canadian dollars.

Table 2

Our preliminary clinical workload scores for children requiring home ventilation

Statistical analysis

Our results included discrete data and also continuous data. Since none of the continuous data sets passed the D'Agostino–Pearson omnibus test for normal distribution, all data sets were managed using non-parametric methods. The values are expressed as median plus IQR and graphically displayed as box and whisker plots. Paired comparisons were made using the Mann–Whitney test. Three or more groups were compared using the Kruskal–Wallis test with Dunn's post hoc test. The correlation between two variables was examined using linear regression (least-squares fit) and the closeness of fit was summarised by Pearson's product-moment coefficient (r).

Results

Over a 9-month period, we enrolled 90 children and their caregivers for the study from our weekly home ventilation clinic (total clinic enrolment is 130 patients). We were able to include a broad range of ages, disease types and socioeconomic backgrounds. Demographic details of patients and caregivers are summarised in tables 1 and 3. The study results are summarised under the following headings.

Table 3

The main characteristics of the principal caregivers enrolled in the study

Adequacy of data collection

The high level of enthusiasm among families for research into home ventilation was reflected in their commitment to the study requirements. Only one family declined to enter the study. The 90 enrolled families complied with all questions in the clinic and every family was successfully contacted by telephone, 1 and 2 months later, for collection of data recorded at home.

Quality of life for ventilated children

Despite challenges, the great majority of children were able to live normal active lives. Of all children, 42.2% were able to answer quality of life questions directly, 57.8% were too young or unable to answer so were assessed by parental proxy. There was no significant difference between the scores produced by the two methods (self-answered PedsQL: median 33.2%, IQR 18.8–49.7% vs parental proxy: median: 35.4%, IQR 25.3–53.5%, p>0.1, Mann–Whitney). Since the two techniques gave comparable results, we combined them for the purposes of analysis and graphical display. Self-reported values are distinguished from proxy values by using open and closed circles (figures 2 and 3). In over 90% of children, the impact of home ventilation was assessed as mild (47.8%) or moderate (45.6%) (figure 1). Over 80% of school-age children attended regular school while the remainder all had some mix of home schooling and partial school attendance. There was a fair linear relationship between the adverse impact of home ventilation on the child and the patient workload score (figure 2), but we found no other significant correlations with social determinants (family income, primate caregiver's education level or length of time on ventilation, all figure 2). Of clinical relevance, there was a positive correlation between the caregiver's quality of life and that of the ventilated child (figure 3).

Figure 1

The adverse effects of home ventilation on affected children (assessed by Paediatric Quality of Life score) and their primary caregivers (assessed by the Caregiver Impact Scale). Both scores are expressed as a percentage of the maximum score. Mild (0–33%), moderate (34–66%) and Severe (67–100%).

Figure 2

The relationship between the ventilated child's quality of life, assessed by the Paediatric Quality of Life score (PedsQL), and four major socioeconomic variables. Closed circles represent PedsQL scores based on proxy answers, and open circles represent PedsQL scores based on self-report. Increasing PedsQL scores represent worsening adverse impacts of home ventilation. Top left: PedsQL versus the patient's workload score, top right: PedsQL versus the child's time on home ventilation, bottom left: PedsQL versus total family income, bottom right: PedsQL versus primary caregiver's education level. Level 1: high school or less, level 2: vocational training or community college, level 3: university, level 4: postgraduate study. Regression lines are calculated using least-squares method. Goodness of fit is expressed as Pearson's product-moment coefficient (r).

Figure 3

The relationship between the ventilated child's quality of life (assessed by the Paediatric Quality of Life score, PedsQL) and the primary caregiver's quality of life (assessed by the Caregiver Impact Scale, CIS). Closed circles represent PedsQL scores based on proxy answers, and open circles represent PedsQL scores based on self-report. In both quality of life scales, increasing scores represent worsening adverse impacts of home ventilation. Separate regression lines for PedsQL values calculated by self-report and proxy are calculated using least-squares method. Goodness of fit is expressed as Pearson's product-moment coefficient (r).

Quality of life for primary caregivers

There was a large gender disparity between caregivers; nearly 90% were women (all but two of whom were the child's biological mother, table 3). The burden of home care was reflected in lower levels of mental and physical health among the primary caregivers compared with their children. Nearly one in every four caregivers reported the adverse effects of home ventilation as severe (figure 1). There were no differences in quality of life scores between male and female caregivers (median CIS 47.4, IQR 24–66 for women vs CIS 52, IQR 38–84 for men). Overall, 53.3% of all caregivers reported health problems that required daily medication (mainly anxiety, depression, hypertension, arthritis) (table 3). The rate of associated illness rose to nearly 90% among those caregivers reporting severe adverse effects of home care (figure 4, top). Caregivers with medical problems reported significantly higher adverse effects of home care compared with those without health problems (figure 4, bottom). There was a fair relationship between the adverse effects of home care on the caregiver and the child's workload score, but there was no correlation with other social variables (figure 5).

Figure 4

Top: the association between the primary caregiver's quality of life, assessed by the Caregiver Impact Scale, (CIS), and the rate of associated chronic health problems. Increasing CIS scores represent worsening adverse impacts of medical condition. Scores for the CIS are divided into mild: 0 to 33% of total, moderate: 34 to 66% of total and severe: 67 to 100% of total. Bottom: the effect of associated chronic illness on the primary caregiver's quality of life, assessed by Caregiver Impact Scale (CIS). Increasing CIS scores represent worsening adverse impacts of home care. Horizontal bar, median; box, 25th–75th centile; whiskers, full range. Asterisk represents significant difference (Mann–Whitney, p<0.01).

Figure 5

The relationship between the primary caregiver's quality of life, assessed by the Caregiver Impact Scale (CIS), and four major socioeconomic variables. Increasing CIS scores represent worsening adverse impacts of medical condition. Top left: CIS versus the patient's workload score, top right: CIS versus the child's time on home ventilation, bottom left: CIS versus total family income, bottom right: CIS versus primary caregiver's education level. Level 1: high school or less, level 2: vocational training or community college, level 3: university, level 4: postgraduate study. Regression lines are calculated using least-squares method. Goodness of fit is expressed as Pearson's product-moment coefficient (r).

Social and economic costs of home ventilation

In British Columbia, funded home care must be provided by fully qualified nurses; there is no alternative level of care. We only collected nursing hours; we did not examine the caregiver's opinion of nursing quality. The median time of government-funded home nursing was 32 h/week but was over 100 h/week for the most complex children (table 4). The primary caregiver's time commitment was still significant. The median value for care time was 4–5 h/day but the spread was large with one family reporting nearly 19 h/day (table 3). Only 24.4% of caregivers were able to work full time; 12.3% were on medical leave. Contrary to common belief, family break-up caused by the stress of homecare was unusual. The divorce rate was 15.6% (table 3). Over the 2-month data collection period, the median value for non-reimbursed costs was below $90/month (table 4). A few families chose to hire more nursing or had non-reimbursed medications so there were outliers but the majority of families did not face severe financial challenges.

Table 4

Time commitment and financial costs associated with home ventilation

Patient workload score

Although the preliminary version of our workload score has not been formally validated, it appears to be a good starting point for future research. There was a good correlation between our workload score and the hours of daily care in the home recorded by the primary caregiver (figure 6). Given the number of variables involved, there was also a fair correlation between workload score and quality of life, both in the child (figure 2, top left) and also in the primary caregiver (figure 5, top left).

Figure 6

The relationship between patient complexity (assessed by patient workload score) and the average time devoted to patient care by the primary caregiver (measured in hours/day). Regression line is calculated using least-squares method. Goodness of fit is expressed as Pearson's product-moment coefficient (r).

Discussion

Since it has been shown that health professionals underestimate the quality of life of children with severe disabilities,23 ,24 it is important that management decisions are based on data obtained directly from the families and children involved.25 Although there are studies aimed at single elements of home ventilation,8 ,26 we could find no research that looked at the impact of managing home ventilation upon the whole family. Consequently, we studied 90 families from a single paediatric home ventilation centre. In addition to measuring the quality of life of the children and their caregivers, we also examined the broader social and economic impacts of home ventilation upon each of the families.

From the ventilated child's point of view, home ventilation appears to be a successful medical strategy. Despite the challenges of daily care, many children who would not have survived three decades ago now lead active and healthy lives. In keeping with the descriptive studies of Noyes14 and Gilgoff,27 the full picture that emerged was extraordinary—overseas holidays, camping trips and participation in wheelchair sports were the norm, not the exception. Based on quantifiable questionnaire data, the adverse effects of home ventilation were scored as mild or moderate in over 90% of the study children—also in line with the findings of Lumeng et al.26 The positive relationship between the quality of life of the child and the primary caregiver (figure 3) has not been described before but it is not surprising since the caregiver is usually also the child's mother. Our experience would suggest this is cause and effect, rather than a simple statistical association. Consequently, we believe that efforts directed towards improving the caregiver's quality of life are also likely to have associated benefits in terms of the child's health and happiness.

Unfortunately, our findings are not so positive when it comes to the primary caregivers—most of whom are women. This obvious gender disparity has received very little research attention.28 In contrast to earlier work by Patterson et al,29 we found no differences in adverse effects between male and female caregivers. In keeping with the findings of Brehaut et al,13 we found that half of all caregivers had chronic health conditions that required daily medication (table 3) and nearly 25% reported that the adverse effects of care were severe (figure 1). In our experience, the reluctance of caregivers to make unprompted complaints about their health, combined with the unpredictability of social cues, makes it very difficult to identify caregivers with serious personal problems. The only reliable way to detect those who need extra help is to meet separately with all caregivers so that each has an opportunity to discuss their personal concerns in private.30 We believe this should be a routine part of every clinic visit.

The ability of families to adapt to the challenges of home ventilation is likely due to a complex mixture of personal factors and past life experiences unique to that group of individuals.31 While we have been able to show that high levels of caregiver stress exist, we were not able to determine the principal contributory causes. With so many variables, it is not surprising that the final quality of life varies widely and is difficult to predict. In contrast to current models of family adaptation to chronic illness,32 we found no relationships between quality of life and annual income, caregiver education or years of home ventilation (figures 2 and 5). The only factor that indicated a greater risk of adverse effects was the child's daily workload (figure 5) but, even there, parents caring for children of the same workload score varied widely in their perception of adverse effects. The only way to be sure that children and caregivers are coping is to include recognition and early intervention for unmet family needs as an integral part of home ventilation management.32

Our service is fortunate that British Columbia's population health policy is based on the concept of strengthening resilience through early positive interventions.33 For the growing number of technology-dependent children, this is reflected in generous government support. Families receive free home nursing by qualified staff plus financial coverage for equipment and medication costs. Of particular note, families also have easy access to respite care in North America's first stand-alone paediatric hospice facility.34 It is hard to imagine how families cope with home ventilation in less supportive environments. Our findings suggest that broader care for the whole family, particularly concentrating on the health of the caregiver, should be an integral part of the management of home-ventilated children. We hope that our results, plus the preliminary workload score, will be of value to others interested in the challenging field of paediatric home ventilation.

References

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Footnotes

  • Contributors KM, AB and AK were responsible for collecting and storing patient data. All listed authors were then equally involved in data analysis and preparation of the final manuscript.

  • Funding The study was funded by the Judi Bowden Memorial Fund for Respiratory Research.

  • Competing interests None declared.

  • Patient consent Obtained.

  • Ethics approval The study was performed in accordance with current research standards of ethics and human rights. The study protocol was reviewed and given formal research approval by the University of British Columbia's research ethics committee. Registration number: H14-00979.

  • Provenance and peer review Not commissioned; externally peer reviewed.

  • Data sharing statement All data from the study are included within this submitted manuscript. There are no unpublished data that are being reserved for other parties. If there are interested readers who wish to have access to the spreadsheets containing the individual data, we are happy to share this information.

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