Parent Perspectives on the Child Experience of Pediatric Home Ventilation via Tracheostomy Free

This study followed the Standards for Reporting Qualitative Research guidelines to improve its transparency and promote trustworthiness.²⁶  This analysis was part of a multicenter project studying the family experience of decision-making regarding pediatric home ventilation using semistructured interviews.¹⁰  English-speaking parents of children, which includes adoptive, foster, and grandparents if they were the primary caretakers and decision-makers for the child, who faced decisions about home ventilation within the previous 5 years were recruited from geographically diverse academic medical centers across 3 states in the South, Northeast, and Northwestern United States. All families were cared for at tertiary hospitals dedicated to pediatric care and had home ventilation programs that each serve between 100 and 200 children annually.

We classified children based on the age of initial respiratory failure, medical complexity, and severe neurologic impairment. For this study, a patient with medical complexity is defined as someone with a combination of multiorgan system involvement from chronic health condition(s), functional limitations, ongoing use of medical technology, and high resource need or use.^27–29  Children with severe neurologic impairment are defined as having “a group of disorders of the central nervous system which arise in childhood, resulting in motor impairment, cognitive impairment, and medical complexity, where much assistance is required with activities of daily living.”³⁰ 

Purposive sampling was used to include a variety of perspectives of study participants who made decisions for or against home ventilation from different community settings (suburban, rural, urban) and geographic regions.³¹  Our team did not explicitly recruit more than 1 parent per child, however, if 2 parents wanted to be interviewed together, their data were analyzed as a family unit. Families were recruited until thematic saturation was achieved; a total of 38 families who made decisions for or against starting home ventilation participated in the larger study. The current analysis focuses on a subset of 21 of the 38 families who chose to pursue home ventilation. The study was approved by the Institutional Review Boards at each institution and informed consent was obtained from each parent.

Qualitative interviews lasting 30 to 120 minutes were conducted via phone and audio-recorded by 1 or 2 members of the local research teams between December 2018 and October 2019. All interviewers were trained to promote consistency in interpretation and implementation of the interview guide that was developed. The semistructured interview guide included domains about the child and family experience of home ventilation suggested by prior research (Appendix).^8,12,32  To limit bias and ensure content relevance, the interview guide was critically reviewed by a study advisory council, which included parents of children who had faced decisions about home ventilation; clinicians in palliative care, complex care, pulmonary, bioethics, and intensive care; and case managers, home healthcare providers, and decision-science researchers. Changes to the guide based on this feedback, were mostly minor edits to clarify wording. Interviews were transcribed and returned to participants for member-checking to enhance trustworthiness of the data.^26,33–36  Transcriptions were then deidentified and stored in password-protected electronic files. Participants received a $50 gift card.

A team consisting of 2 pediatric pulmonary physicians and bioethicists (H.V. and B.W.), one of which has extensive experience in qualitative analysis (B.W.), and a trained research coordinator (A.M.) conducted conventional content analysis on deidentified transcripts limited to portions related to the child’s respiratory insufficiency and experience of home ventilation. Interview transcripts were read to achieve immersion and obtain a sense of the whole, then emerging codes were organized into an initial coding scheme based on participants’ perspectives that was grounded in the data.^37,38  Through an iterative process, inductively generated codes were organized into comprehensive coding structures using Dedoose software.^37,39  To further refine the coding schema, this was then reviewed by the full study team, which included 2 critical care physicians and bioethicists (R.B., C.H.), one of which is also a palliative care provider and an expert in qualitative analysis (R.B.), and a pediatric pulmonary physician and bioethicist (N.J.). The remaining transcripts were then jointly coded by 2 authors (H.V., A.M.) utilizing a final codebook. Unresolved coding discrepancies were reviewed by a third reviewer (B.W.). Following completion of coding, regular discussions were held to discuss findings and identify themes with the full study team. Several themes were consolidated into each domain that summarized and represented the data. These themes were then organized into subthemes of whether that aspect of the child’s experience improved or worsened with home ventilation.

Twenty-one family units (of either 1 or 2 parents) contributed data to this analysis and represented various social contexts, including geographic regions, education levels, and household incomes (Table 1). Sixteen children (76%) experienced respiratory failure in the first year of life, 17 (81%) had medical complexity, and 11 (52%) had severe neurologic impairment related to congenital conditions, chromosomal differences, or traumatic injury (Table 1).

The child experience of home ventilation fell into 5 domains, as described by their parents: (1) health and well-being; (2) development; (3) adaptation; (4) mobility and travel; and (5) relationships. Figure 1 demonstrates how each domain is oriented across a continuum of whether the child’s experience generally improved or worsened according to their parents.

Twenty out of 21 families described improvements to their child’s overall health and well-being, which included the child’s respiratory health, daily care regimen, and prolongation of life (Table 2). Improvements in their child’s respiratory health was demonstrated by increased comfort, improved work of breathing, fewer respiratory illnesses, and decreased need for supplemental oxygen following initiation of home ventilation. These benefits allowed them to “act more like a baby (or child), not working so hard to breathe” (Family 1). There were 2 families, however, that identified worsened respiratory health with frequent respiratory interventions as they transitioned home (which subsided over time) and more respiratory illnesses as their lung disease progressed.

Six families described that their child had a lower daily care regimen burden following initiation of home ventilation, which were described as the child having fewer medical procedures, decreased need for medications, improved comfort in clearing secretions, and improved tolerance of illnesses. However, 14 families described that their child’s days involved substantial time devoted to medical care for home ventilation, including numerous medical appointments and frequent respiratory treatments or medications.

All families identified that their child’s life was prolonged because of home ventilation. However, 1 family described regret in their choice to prolong their child’s life because of “the amount of interventions and things that are painful that have to be done to her on a near constant basis,” such as airway clearance treatments and blood draws (Family 12). This family described that though home ventilation prolonged her child’s life, it did “not actually help her live” (Family 12).

Thirteen families described overall improvements, whereas 6 families described overall declines in their child’s development (Table 3). Thirteen families expressed that their child thrived developmentally and was able to “grow” and “progress” following home ventilation. Ten families specifically saw improvements in their child’s physical development with descriptions of better growth, increased energy and activity, and quicker attainment of milestones when compared with the time before home ventilation. There was 1 family that said their child’s physical development diminished following home ventilation, with a decreased growth trajectory, slower attainment of milestones, and less engagement in activities. These developmental improvements and declines may have been because of home ventilation and/or the expected progression of the child’s underlying condition.

Only half of parents commented on their child’s speech development. Of these, parents were relatively split on whether their child’s speech development was improved or hindered following home ventilation. Three families described that their child was able to speak louder and more clearly, whereas 5 families described that their child was unable to speak or cry following home ventilation. For example, 1 parent remarked that her child was not strong enough to “push her voice out around the trach” (Family 21) and another parent detailed how they couldn’t hear their child crying over the ventilator (Family 1).

For the domain of the child’s adaptation to home ventilation, 13 families described that the child’s experience of the home ventilation medical equipment seemed routine, natural, and “part of their child” (Table 3). One family described how her child “loves the trach. She doesn’t mind it at all… she can breathe easy, so she’s happy with it” (Family 6). Four families expressed that their child did not like home ventilation, with descriptions that their child “really struggled accepting the ventilator” and “thinks it’s weird.” Family 11 described how their child was constantly “yanking on” the cords and was “irritated by them.”

Fifteen families shared that their child appeared to be more comfortable and happier following home ventilation with descriptions of the child “laughing more” and being more “content” than they were before home ventilation. Two families reported that their child was less happy and constantly “fighting the ventilator” or trying to “pull off the cords.” Although 13 families described that home ventilation permitted their child to pursue a “new normal,” such as allowing the opportunity to “be creative, contribute to society, (and) act like a regular child,” 3 families described how home ventilation prohibited the pursuit of a “normal life” by keeping them from participating in daily activities that their peers were involved in, such as sports.

Home ventilation’s impact on the child’s mobility and ability to travel was the only domain where families consistently described negative experiences overall (Table 4). Seventeen families reported that their child’s mobility was restricted, with descriptions that home ventilation kept their child “tied down” because of all the cords and machinery. However, 16 families placed significant value on home ventilation providing the child with enough mobility to leave the hospital, allowing the child to “make new memories,” and experience a better quality of life away from the hospital. Six families described that being home is what allowed their child to really “thrive.”

The travel experience for children was typically described as cumbersome, which initially decreased parents’ ability and/or desire to take the child outside the home. One family described how traveling with the child meant “taking the whole house with her,” therefore she “didn’t really take him anywhere” (Family 13). Fifteen families discussed that although travel was initially scary or daunting, it often became smoother with time and eventually “second nature.” Family 4 described how “at first, it’s scary… but once we got familiar with it, it was the most freeing thing in the world, and it was so cool because she got to experience all these things.” Another family described how her child “gets so excited” whenever she was placed in her car seat and how she “loves going on trips” (Family 9).

Twenty families felt home ventilation improved their child’s capacity to form relationships, including more play with their parents and siblings (Table 4). One family described how happy her child “was to be around her siblings again and very happy to be in the surroundings that she was used to” with her family (Family 7). Families conveyed that their family unit became more cohesive, and the child was more incorporated into the family following home ventilation since they were able to participate in family activities to create new memories. These families described that their child participates in most of the things they were able to with their family before home ventilation.

However, relationships with others outside of the home were often harder to achieve. Because the child was perceived as fragile, this resulted in less interactions with others and less integration into the extended family and community. Family 2 describes how for her child, “her relationship is kind of hard with others because some people are scared to talk and come around her, they’re scared they might hurt her.” Relationships with others were impacted by concerns that the child would contract an illness or be uncomfortable given their decreased mobility, amount of equipment, and overall appearance. Family 11 described how the “more and more machines [child’s name] got, the less family came around.”

In this study, 21 families with children who used home ventilation described both positive and negative aspects of their child’s experience of the technology, however, descriptions of these domains were generally positive (Fig 1). The most consistently positive child experiences of home ventilation reported by families were overall development and improved comfort, happiness, and play. The most consistently negative experience was home ventilation’s impact on the child’s mobility and ability to travel, which affected the frequency that the child could be outside the home. The remainder of the experiences were generally mixed: improved overall health and well-being following home ventilation but only with intensive daily care, and more integration into family activities following home ventilation but difficulty forming relationships with those outside the home.

Families have reported feeling unprepared for changes when they were not informed of both the potential positive and negative consequences of home ventilation.^9,22,24,25  Even though a child’s needs for home ventilation and the trajectory of their health following initiation of home ventilation are unique, the 5 domains of the child experience identified in this study can provide a framework for clinicians in the inpatient setting to help prepare families for what to expect. Additionally, many families choose to either pursue or decline interventions based on their perspective of how it may impact their child’s quality of life, however quality of life may be difficult to define and may mean different things for different families. This framework with the 5 domains can be used by clinicians to help families consider the positive and negative impact that home ventilation may have on certain aspects of their child’s quality of life. For example, although our study found that most children benefited from improvements in their overall development, there was an element of isolation present throughout various domains as they often had limited mobility, decreased verbalization, and fewer interactions with others following home ventilation. Providing balanced counseling of both positive and negative child experiences can give families more realistic expectations and help avoid an optimistic or pessimistic bias on the part of the clinician and family as they engage in the decision-making process.

Factors such as the child’s underlying medical condition(s), familial values, and social context affect how families prioritize certain domains over others when deciding whether to initiate home ventilation.¹⁰  For decisions where it is unclear whether there will be benefit or harm to the child, it is important for parents to appreciate that there is no right or wrong decision, only the best decision that could be made for their child.⁴⁰  In the United States, this decision falls within the zone of parental discretion, which represents the limits within which parental values are permitted to shape decisions.^41,42  By providing families with information on what they could expect for their child, they can better understand whether home ventilation would serve their child’s interests in the context of their values and goals.⁴³ 

Since this information can support a family’s decision-making process, it would be valuable for inpatient clinicians to understand the nuances of the child’s long-term experiences of home ventilation from experienced families.^11,25  In the past 2 decades, several studies described the impact of home ventilation on children’s physical health, social experience, sense of autonomy, comfort, and overall quality of life.^{12–16,44,45}  However, many of these prior studies had a mixture of children who used noninvasive and invasive home ventilation, were school-aged or adolescents, lived long enough to participate in the studies, and/or had the neurodevelopmental capacity to answer interview questions. Our data add to the clinician’s toolkit by providing information on various domains of the child experience of home ventilation through firsthand perspectives of families with children whose ages and neurodevelopmental disabilities reflect the broader group of children using pediatric home ventilation in the United States, as well as the growing number of children with medical complexity.^17–21  This information can be used by clinicians to improve the decision-making process for families facing this decision by helping to facilitate meaningful conversations that describe the balanced advantages and disadvantages of home ventilation, a strategy that was identified by this cohort of parents.⁷ 

There are several limitations to this study. First, these descriptions may have recall bias as the interviews took place up to 5 years following initiation of home ventilation. However, it was important for enough time to have lapsed to have long-term experiences to describe. Second, although thematic saturation was achieved for this analysis, recruitment for this study was done via purposive sampling, which may affect the generalizability of our findings. Third, our cohort reflects a broad range of clinical conditions leading to tracheostomy being recommended for the child. Focusing future research on a cohort with specific indications for tracheostomy, such as chronic lung disease of prematurity or progressive neurodevelopmental conditions, may allow for more tailored conclusions. Fourth, the study does not address the experiences of children living in chronic care facilities. Future studies to improve understanding of these experiences from families and chronic care facility providers would enhance clinician understanding in various outpatient settings. Lastly, although study participants had geographic, educational, and economic diversity, there was limited racial and ethnic representation. There were also no parents who had a preferred language other than English. Increased understanding of the experiences of families from diverse racial and ethnic backgrounds as well as preferred languages is important for future studies given the disparate outcomes for these patient populations.^46–50 

The decision to initiate pediatric home ventilation is one of the most complex that parents make. There are various domains of the child’s experience that both improve and worsen following initiation of home ventilation. Clinicians who work in the inpatient setting guide families through these decisions and ought to understand what other children have previously experienced. Understanding these balanced experiences can improve clinician counseling so that future families facing the decision of home ventilation may benefit from the perspectives of families with the experience and expertise in caring for children on home ventilation.

We thank Jessica Raisanen and Kelly Shipman who contributed to the development of the interview guide, family recruitment, and conducting interviews; Jenny Shepard, who is the project manager for our ongoing multi-institution collaboration, for her support; and the families who participated in the interviews.