The Queensland paediatric tracheostomy quality of life study

Introduction

Currently, there is no Australian data on the quality of life (QOL) of families and/or caregivers of a child living with a tracheostomy. The published QOL literature utilising The PedsQL^TM Family Impact Module Version 2.0 (a validated QOL assessment) is from the United States of America (USA), United Kingdom (UK) and India (1-3). In Australia, paediatric tracheostomy is not National Disability Insurance Scheme (NDIS) eligible due to the perpetual potential for the tracheostomy to be removed. Further, there is a reluctance to label a child as having a disability purely due to a tracheostomy. However, in reality, caring for a child with a tracheostomy requires additional human (24 hours a day, 7 days a week) and financial resources that are otherwise not recognised by any formal support package in Australia.

In contrast, at Great Ormond Street Hospital in London, UK, the National Health Service (NHS) provides carers, when available, to support families, offering an estimated average 40–50 hours per week, typically covering 4 to 5 full nights of care. Some single parent families in the UK are reported to receive 7 nights of respite. In the USA, eligibility for home nursing depends on the child’s insurance and staff availability (4). USA health insurance companies have case managers to assist families to navigate the health system (4). At the University of California Davis Children’s Hospital in Sacramento, USA, families living with a child with a tracheostomy are supported by a tracheostomy clinic (4). The hospital has an In-Home Support Service (IHSS) which helps fund services so the child can remain in their home (4). In India, there is no funding support for families living with a child with a tracheostomy. Families often recycle equipment to minimise costs. Health insurance only covers the cost of hospitalisation. However, many households have extended families under the one roof which provides a pool of potential carers who may be home full time to permit parent caregivers to work.

Given these differences between the USA, UK, India and Australian paediatric tracheostomy carer support in place, it raised the question of whether the current QOL data in publication was applicable to Australia. The overseas data to date has highlighted a profound issue. Caregivers of children with a tracheostomy are challenged to maintain competency in management of this medical technology that may be accompanied by a child with complex additional medical needs. They may not have any healthcare background or support network. They may have limited physical and/or financial resources. Without local data, it is possible that the current burden on caregivers of Australian children with a tracheostomy is underestimated.

The aim of this study was to collect Australian data on the QOL outcomes for caregivers of paediatric tracheostomy to better understand the local QOL challenges.

Methods

A prospective cohort study was undertaken utilising two questionnaires. The PedsQL^TM Family Impact Module Version 2.0 and a second questionnaire on access to respite (provision of a subsidised tracheostomy carer) and a psychologist. The study is reported according to the STROBE reporting guidelines (available at https://www.theajo.com/article/view/10.21037/ajo-24-69/rc).

The PedsQL^TM Family Impact Module Version 2.0 is a 36-item questionnaire that measures parent self-reported functioning across six domains: physical, emotional, social, cognitive, communication, worry as well as two scales measuring family functioning: daily activities and family relationships (5). The survey can be self-administered or done by proxy (1). A 5-point response scale is used (0 = never a problem; 4 = always a problem), then items are reverse-scored and linearly transformed to a 0–100 scale so that higher scores indicate better functioning (5). Scores are reported as means and standard deviations. The PedsQL^TM Total Family Impact Score is the sum of all 36 items divided by the number of items answered. Families with otherwise healthy children average scores in the 80s (1). The Parent Health-Related Quality of Life (HRQOL) Summary Score is the sum of the 20 items answered in the physical, emotional, social and cognitive function scales (5). The Family Functioning Summary Score is the sum of the eight items in daily activities and family relationships divided by the number of items answered (5).

A second 4-item questionnaire compiled by the authors was also administered to participants that sought information on respite care (access, number of hours per week, perceived utility), access to a psychologist (and perceived utility) and to the NDIS (see Figure 1).

Figure 1 Second tracheostomy carer quality of life questionnaire in the Queensland paediatric tracheostomy quality of life study.

All families of children (inpatient and outpatient) currently living with a tracheostomy and receiving care with the Department of Otolaryngology at the Queensland Children’s Hospital (QCH) were invited to participate during July 2023. Parental informed consent was obtained. The following demographic data was collected where a non-anonymised survey was returned: age of child, gender, tracheostomy indication, tracheostomy duration, use of home ventilation and remoteness of residential address (metropolitan, rural or remote) (6).

PedsQL^TM Family Impact Module Version 2.0 scores were compared to published scores from similar overseas study of cohorts of caregivers of children with a tracheostomy (1-3) as well as a cohort of medically fragile children (children with chronic health conditions including cerebral palsy and birth defects) requiring home or nursing home placement in San Diego (5). The Australian and overseas PedsQL^TM Family Impact Module Version 2.0 mean scores were subjected to an independent t-test to investigate for significant differences (or Mann-Whitney U test when the data did not follow a normal distribution).

The study was conducted in accordance with the Declaration of Helsinki and its subsequent amendments. The study was approved by the Children’s Health Queensland Hospital and Health Service Human Research Ethics Committee (HREC/23/QCHQ/97325) and informed consent was taken from all individual participants.

Results

Eighteen of 42 (43%) families participated in the study. Seven children were female (39%). The primary indication for tracheostomy was upper airway obstruction (n=13 or 72%) whereas 5 required ventilation (28%). There was a range of pathology including lymphatic malformation, teratoma, subglottic stenosis, bilateral vocal cord paresis, severe laryngomalacia, Pierre Robin Sequence, Velocardiofacial syndrome, Cri du chat and congenital central hypoventilation syndrome. Ten families (56%) were located in a metropolitan zone, 6 rural (33%) and 2 remote (11%) (see Table 1).

Seventeen of 18 (94%) respondents reported NDIS access. Two of 18 (11%) were supported by the Home Ventilation Service at the time of survey. Seven of 18 (39%) families reported access to respite ranging 9 to 28 hours per week (median 10 hours). One caregiver responded they never received the full respite in their plan due to issues finding support workers. Six of 7 (86%) recipients of respite reported this as useful. One respondent (14%) felt respite was “sometimes” useful. They detailed how they previously worked while their carer was present however subsequently carers could not start early enough. Five of 18 (28%) families reported access to a psychologist. However, 11 of 18 (61%) felt that a session with a psychologist would be useful for the family.

Results from the PedsQL^TM Family Impact Module Volume 2.0 are detailed hereafter. Firstly, the mean Total Family Impact Score was 44±20 (range, 14–90) which is below the average score (80s) for otherwise healthy children (1). It is also lower than scores from international cohorts, including the Dallas cohort (76±19), New Delhi (55±16), Sheffield (62±19), the San Diego cohort of medically fragile children in outpatient (63±17) and inpatient care (81±17) (1,5). Within this study, the mean Parent HRQOL was 44±20 (range, 10–89) and the mean Family Function Summary Score was 45±23 (range, 6–97) (see Table 2).

The 3 lowest (worst functioning) mean domain scores were:

• Family daily activities: 25±29;
• Worry: 36±26;
• Social function: 40±25.

The 6 lowest (worst functioning) individual question scores were:

• “Family activities take more time and effort”: 18±27;
• “I worry about my child’s future”: 21±27;
• “I feel tired during the day” AND
• “It is hard to find time for social activities”: 28±27;
• “I have difficulty finding time to finish household tasks” AND
• “I feel too tired to finish household tasks”: 29±35.

The 3 highest functioning domains were:

• Family relationships: 58±23;
• Emotional function: 48±26;
• Communication: 46±31.

In comparison to the overseas QOL literature utilising The PedsQL^TM Family Impact Module Version 2.0 from the USA, UK and India (1-3), the Queensland cohort scored the worst mean Total Family Impact Score (see Table 3).

When comparing the mean Total Family Impact PedsQL^TM Family Impact Module Version 2.0 score to the San Diego [2004] (5) group of medically fragile children in outpatient care, the Australian cohort scored worse (mean =44 vs. 63). The largest difference between the mean scores was in cognitive functioning. Further comparing the San Diego cohort of medically fragile children in inpatient care (5), the mean PedsQL^TM Family Impact Module Version 2.0 Total Family Impact Score was still worse for our Australian cohort (mean =44 vs. 81). The largest difference in mean scores was family daily activities (mean difference =60).

Highly significant differences were found between Queensland and all overseas cohorts (P<0.001). There was a significant difference between the Queensland and San Diego outpatient cohort [mean difference; −19.5, 95% confidence interval (CI): −27 to −12, P<0.001]; Queensland and San Diego inpatient cohort (mean difference: −38.4, 95% CI: −45 to −32, P<0.001); Queensland and Dallas (mean difference: −32, 95% CI: −38 to −26, P<0.001); Queensland and New Delhi (Mann-Whitney U statistic 5, P<0.001, rank biserial correlation effect size 0.917) and Queensland and Sheffield (mean difference −18.7, 95% CI: −27 to −11, P<0.001).

Within our Australian data, when comparing the domain scores to each other, the largest difference between the means was family daily activities vs. family relationships (favoured) (difference of means =33).

Discussion

The findings of this initial Australian study into the QOL of families of children living with a tracheostomy, like the overseas literature, reports a quality-of-life burden.

In 2019, authors from Sheffield published PedsQL^TM Family Impact Module data from 25 families (3). The total family impact score was 61.6 (higher than our mean score of 44) (3). The authors are aware of the availability of more hours of tracheostomy carer support (albeit at times met with difficulty finding suitable carers) at the Great Ormond Street Hospital in the UK, compared to our Queensland families. While this may not be the only reason for the difference in the QOL score between the surveyed UK versus Australian caregivers, it is a disparity in funding for families.

A group from Dallas also published a PedsQL^TM Family Impact Module questionnaire-based study in 2021 of 98 families of a child with a tracheostomy aiming to measure QOL among families of children with a tracheostomy (1). The total family impact score was 76 (compared to our score of 44) (1). The authors are also aware of differences in funding and clinical support offered in some USA institutions compared to ours such as dedicated tracheostomy clinics, avenues for home nursing and health insurance provider eligibility criteria (4). Similarly to the Sheffield data (3), the lowest domain score was also family daily activity problems (mean score 67).

Family daily activities was also the worst rated domain in our Australian data. Whereas greater access to tracheostomy respite would assist, so would access to community resources such as housekeeping, gardening and grocery delivery. It is interesting that family relationships are one of the most highly rated domains (1,3). Perhaps the shared experience of adversity among family members, with time and effort rebounds to resilience. This was noted by Huang et al. [2023] in their publication on Chinese families of children with leukaemia (7). The authors referenced how some families could endure the worst suffering and rebound from adversity stronger (7). The authors noted a two-stage process by which families respond to illness and transition from disruption (informed by negative emotional disturbances and the challenges of the diagnosis and treatment) to cultivating resilience by increasing positive attitudes, establishment of family routines, activating support systems and employing open family communication (7). Clinicians may benefit from being aware of this process of developing family resilience and supporting resilience-focused strategies.

Comparing the overseas data based on the PedsQL^TM Family Impact Module to study paediatric tracheostomy caregiver QOL, our Queensland participants rated themselves worse than overseas cohorts and worse than parents of medically fragile children. The largest difference in the mean scores versus the medically fragile children in outpatient care was cognitive function. Clinicians are in a position to lessen cognitive burden by providing more support in tracheostomy cares. Currently, Australia does not have a standardised paediatric tracheostomy educational package, and this would be beneficial to provide families with a comprehensive resource that is consistent between Australian institutions. In this institution, a dedicated outpatient tracheostomy nurse is not funded. However, this would assist the burden on caregivers via either home visits, telephone support or other regular follow-up. A monthly tracheostomy multidisciplinary team (MDT) clinic would optimise access and communication with (and among) the various healthcare practitioners supporting caregiver families. This might be comprised of a core team of: Ear Nose and Throat surgeons, clinical nurse specialists, respiratory physicians, speech pathology, physiotherapy, dietetics and social work meeting with the child and carer/s in the outpatient clinic. In addition, it would benefit from collaboration with other hospital services on a case-by-case basis (perhaps by invitation in anticipation of a given patient’s expected attendance in an upcoming MDT clinic) such as (but not limited to) intensive care, occupational therapy, cultural liaison and paediatric medicine. The literature cites MDTs can offer better coordinated management (8,9).

The other aspect to comparatively worse cognitive burden than parents of medically fragile children may be psychosocial support. Among our cohort, a minority (28%) had accessed a psychologist. Formal psychological preparation of parents and/or assessment is not routine in our institution. Further, even if it were, it may not be acceptable to all due to the stigma associated with mental health care. Yet the stress may be immense with the hospital-to-home transition, particularly for families without home nursing support and the prospect of newly managing airway complications. Then there is the burden of the change in daily routine, changes to financial resources due to loss of paid work and less time spent in social interactions due to carer responsibilities. There is the burden of the uncertainty of the impact of a tracheostomy on their child’s and the parents’ future/s. In Koch and Jones’ 2018 paper, they reference a consensus on the topic of parent caregiver psychosocial assessment in paediatric cancer (10). They highlighted the understanding that care for the parent impacts on the child (10). The authors concluded that the treating team should provide psychosocial assessment and evidence-based intervention, organised respite and attention to distress for all family caregivers to meet goals for caring for the child (10). Future research would clarify the benefit of psychosocial assessment for caregivers of children with a tracheostomy.

In contrast to overseas cohorts, Australian families and patients have access to the NDIS, a financial resource which intends to provide eligible Australians with the support to be “more independent and engage socially and economically” (11). Eligibility includes the presence of a permanent disability that significantly affects ability to take part in everyday activities; age under 65 years old on first application and Australian residency, citizenship or holder of a permanent visa or protected special category visa (11). In our cohort, the majority had NDIS support. The details of this NDIS support are determined on a case-by-case basis, and in some instances may include tracheostomy care in the funding package (however, subject to annual review with the possibility of cessation of tracheostomy funding support). Despite the NDIS facility in Australia, participants reported below average QOL (1-3). Caregivers’ families have expressed frustration that the scheme is tailored to adult individuals rather than families and children (12).

The limitations of this study include a small sample size for this survey-based study. A larger sample would raise confidence for representative data. When the sample size is small, the overall result may be unduly influenced by a few outliers who may hold a particular grievance (justified or not) with the care provided. Further, this study captures a moment in time that may be influenced by proximity to the COVID pandemic. Unfortunately, the study did not collect data on cultural diversity nor the number of fathers versus mothers completing the survey, their respective percentage of caregiving or the family home composition that may also impact scoring.

Conclusions

This is the first study to publish Australian data regarding paediatric tracheostomy QOL. Within this Queensland cohort, the majority of respondents had NDIS access and reported respite to be useful. A minority had access to a psychologist, but the majority felt this would be useful for their family. Similar to overseas data, it was found that caring for a paediatric tracheostomy depresses the caregivers’ QOL. The Queensland cohort returned QOL scores worse than other medically fragile children and children with a tracheostomy from overseas.

Acknowledgments

The authors appreciate Dr. Kamran Syed for his contribution regarding paediatric tracheostomy care in India and Associate Professor Mark Jones who provided statistical support. The authors acknowledge the PedsQL^TM Family Impact Module Version 2.0, contact information and permission to use: Mapi Research Trust, Lyon France, http://eprovide.mapi-trust.org. Our abstract has been accepted for presentation at the 74^th Annual Scientific Meeting for the Australasian Society of Head and Neck Surgery and concurrent Otorhinolaryngology Head & Neck Nurses Group Meeting, Perth, Australia, taken place between 8^th to 10^th March 2024.

Footnote

Reporting Checklist: The authors have completed the STROBE reporting checklist. Available at https://www.theajo.com/article/view/10.21037/ajo-24-69/rc

Data Sharing Statement: Available at https://www.theajo.com/article/view/10.21037/ajo-24-69/dss

Peer Review File: Available at https://www.theajo.com/article/view/10.21037/ajo-24-69/prf

Funding: None.

Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at https://www.theajo.com/article/view/10.21037/ajo-24-69/coif). H.B. serves as Editor-in-Chief of the Australian Journal of Otolaryngology from April 2024 to December 2029. H.B. declared Smith Medical provides sponsorship for the Brisbane Paediatric Airway Course by provision of equipment (tracheostomies) for the workshop. The other authors have no conflicts of interest to declare.

Ethical Statement: The authors are accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. The research was conducted in accordance with the Declaration of Helsinki and its subsequent amendments. The study was approved by the Children’s Health Queensland Hospital and Health Service Human Research Ethics Committee (HREC/23/QCHQ/97325) and informed consent was taken from all individual participants.

Open Access Statement: This is an Open Access article distributed in accordance with the Creative Commons Attribution-NonCommercial-NoDerivs 4.0 International License (CC BY-NC-ND 4.0), which permits the non-commercial replication and distribution of the article with the strict proviso that no changes or edits are made and the original work is properly cited (including links to both the formal publication through the relevant DOI and the license). See: https://creativecommons.org/licenses/by-nc-nd/4.0/.

References

1. Johnson RF, Brown A, Brooks R. The Family Impact of Having a Child with a Tracheostomy. Laryngoscope 2021;131:911-5. [Crossref] [PubMed]
2. Chandran A, Sikka K, Thakar A, et al. The impact of pediatric tracheostomy on the quality of life of caregivers. Int J Pediatr Otorhinolaryngol 2021;149:110854. [Crossref] [PubMed]
3. Westwood EL, Hutchins JV, Thevasagayam R. Quality of life in paediatric tracheostomy patients and their caregivers - A cross-sectional study. Int J Pediatr Otorhinolaryngol 2019;127:109606. [Crossref] [PubMed]
4. Olarewaju A. Pediatric Tracheostomy Handbook: UC Davis; 2019. Available online: https://health.ucdavis.edu/media-resources/children/documents/patient-education-A-to-Z/peds-tracheostomy-handbook.pdf
5. Varni JW, Sherman SA, Burwinkle TM, et al. The PedsQL Family Impact Module: preliminary reliability and validity. Health Qual Life Outcomes 2004;2:55. [Crossref] [PubMed]
6. Rural, Remote and Metropolitan Area: Australian Government Department of Health and Aged Care. Available online: https://www.health.gov.au/topics/rural-health-workforce/classifications/rrma
7. Huang Y, Chen M, Zhang Y, et al. Finding family resilience in adversity: A grounded theory of families with children diagnosed with leukaemia. J Clin Nurs 2023;32:5160-72. [Crossref] [PubMed]
8. Bonvento B, Wallace S, Lynch J, et al. Role of the multidisciplinary team in the care of the tracheostomy patient. J Multidiscip Healthc 2017;10:391-8. [Crossref] [PubMed]
9. Cetto R, Arora A, Hettige R, et al. Improving tracheostomy care: a prospective study of the multidisciplinary approach. Clin Otolaryngol 2011;36:482-8. [Crossref] [PubMed]
10. Koch KD, Jones BL. Supporting Parent Caregivers of Children with Life-Limiting Illness. Children (Basel) 2018;5:85. [Crossref] [PubMed]
11. National Disability Insurance Scheme (NDIS): The National Disability Insurace Agency; 2019. Available online: https://www.ndis.gov.au
12. D'Arcy E, Burnett T, Capstick E, et al. The Well-being and Support Needs of Australian Caregivers of Neurodiverse Children. J Autism Dev Disord 2024;54:1857-69. [Crossref] [PubMed]