The incidence, as well as the predictors of mortality, for children receiving home mechanical ventilation (HMV) using population-based data in Canada is a current knowledge gap. Our objectives were to describe HMV incidence and mortality rates, and associations of demographic and clinical variables on mortality.
Using Ontario health and demographic administrative databases, we conducted a retrospective cohort study (April 1, 2003–March 31, 2017) of children aged 0 to 17 years receiving HMV via invasive mechanical ventilation and noninvasive ventilation. We identified children with complex chronic conditions. We used data from Census Canada to calculate incidence rates and Cox proportional hazards modeling to assess for predictors of mortality.
We identified 906 children with a mean (SD) crude incidence rate of 2.4 (0.6) per 100 000 for pediatric HMV approvals that increased by 37% over the 14-year study period. Compared with children who were invasively ventilated, we found mortality was associated with noninvasive ventilation (adjusted hazard ratio [aHR], 1.9; 95% confidence interval [CI], 1.3–2.8). Mortality was highest in children from families in the lowest income quintile (aHR, 2.5; 95% CI, 1.5–4.0), those with neurologic impairment complex chronic conditions (aHR, 2.9; 95% CI, 1.4–6.4), those aged 11 to 17 years at HMV initiation (aHR, 1.5; 95% CI, 1.1–2.0), and those with higher health care costs in the 1 year before HMV initiation (aHR, 1.5; 95% CI, 1.3–1.7).
The incidence of children receiving HMV increased substantially over the 14-year period. Demographic variables associated with increased mortality were identified, suggesting areas requiring greater attention for care providers.
Home mechanical ventilation (HMV) is an advanced medical therapy offered to children with medical complexity. Despite the growing population of pediatric HMV, the existing literature is limited with regard to sociodemographic factors that may affect mortality in this population.
Fourteen-year data from Ontario confirmed increasing incidence of pediatric HMV. Mortality in children receiving HMV was affected by their underlying medical condition, household income, and health care utilization before HMV initiation, highlighting areas that require greater attention for care providers.
Home mechanical ventilation (HMV) is 1 of the most advanced and complex long-term therapies offered to pediatric patients outside of the hospital setting. Regional and national registries have described significant growth in the population of children using HMV in the past several decades.1–7 This growth has been associated with a paradigm shift from long-term institutional care to community living with their families. Supporting these children at home requires substantial human health care resources given their complex care needs. Furthermore, there is a significant risk of morbidity and mortality given their medical fragility. In the United States, children with complex chronic conditions (CCC) using HMV have inpatient mortality rates that are fourfold higher than CCCs not using HMV.8 Overall mortality rates in children requiring HMV have been reported to be as high as 43%.2,7,9–26 Further, family caregivers of children using HMV face significant financial stress given the need for 1 parent to be an unpaid, full-time primary caregiver, in addition to the significant out-of-pocket costs related to the purchase of needed equipment.27 However, the role of neighborhood income and other demographic and clinical factors on the mortality rate of children requiring HMV at a population level remains largely unknown.
Despite the growing population of pediatric HMV, there is limited evidence on change in survival and mortality rates over time that also enables analysis of risk factors that include socioeconomic status. Our study aims were to:
to describe the incidence and mortality rates in the pediatric HMV population over a 14-year period in Ontario, Canada; and
to identify demographic and clinical variables associated with mortality.
Identification of predictors of mortality is paramount because it offers an opportunity to provide additional support and resources to those children and families at highest risk.
Methods
Study Description
We conducted a retrospective, longitudinal, population-based cohort study from April 1, 2003, to March 31, 2017, using health administrative databases for the province of Ontario, Canada, held at ICES (formerly the Institute for Clinical Evaluative Sciences) (www.ices.on.ca). According to the 2021 Census, the population of Ontario is 14.2 million.28 ICES is an independent, nonprofit research institute whose legal status under Ontario’s health information privacy law allows it to collect and analyze health care and demographic data, without consent, for health system evaluation and improvement. We used unique encoded identifiers to link databases. These databases contain anonymized data for all Ontario residents. Universal public health insurance funded by the provincial government covers the costs of all medically necessary care. The Ontario Ventilator Equipment Pool funded by the Ontario Ministry of Health is a central depot which loans ventilators and associated equipment at no cost to people requiring HMV approved by the provincial Assistive Devices Program (ADP).29 All children using HMV receive clinical care from a hospital-affiliated HMV program. Children receiving invasive mechanical ventilation (IMV) via tracheostomy are eligible for home care nursing. Children receiving noninvasive ventilation (NIV) may receive home care nursing determined on a case-by-case basis. This study is reported in accordance with the REporting of Studies Conducted Using Observational Routinely-Collected Health Data statement.30
Study Participants
We identified a cohort of children with public medical insurance, aged between 0 and 17 years, who had applied for an HMV ventilator (NIV or IMV) from the ADP between April 1, 2003, and March 31, 2017. To capture children who were receiving invasive ventilation via tracheostomy, we used the corresponding medical billing procedural codes. Children were excluded if:
they had an invalid or absent Ontario Health Insurance Plan number;
age was missing from the Ontario Registered Persons Database; or
death was recorded before date of approval of a ventilator.
In cases where multiple ventilator request records were noted, we restricted inclusion to the first record for that individual (assumed to be the first/incident application). Multiple ventilator requests would be present if a child switched ventilators as they transitioned ventilation type.
Data Sources
The ADP database contains all approvals for respiratory-related technologies, such as ventilators and supplies, from 2001 onward. We linked the study cohort to the following population-based administrative databases:
Canadian Institute for Health Information Discharge Abstract Database to identify hospitalizations and demographics, diagnostic and procedural data, length of stay, and in-hospital death;
National Ambulatory Care Reporting System to identify emergency department presentations and ambulatory care visits;
Ontario Health Insurance Plan billing database to identify physician and procedure billing;
Ontario Registered Persons Database for death outside the hospital;
Ontario Home Care database for homecare services; and
Ontario Drug Benefits database.
Children are eligible for Ontario drug benefits if they receive benefits under the Ontario Disability Support Program or professional services under the Home Care Program.31 The International Classification of Disease, 10th Revision-Canadian Codes, were used to establish most responsible diagnosis. We used the pediatric CCC classification system version 2 updated for International Classification of Disease, 10th Revision, codes to classify children with CCCs as neurologic impairment only, another single organ system, or multiple organ systems.32 Feudtner et al defined CCCs as any medical condition that can be reasonably expected to last at least 12 months (unless death occurs) and severely affects either 1 organ system or multiple different organ systems enough to require specialty pediatric care.32
Cohort Characteristics
For all included children, we determined age and sex at the time the application for HMV was approved. Neighborhood income and urban/rural place of residence were ascertained using postal codes linking to Statistics Canada as described previously.33 We documented the following public health service utilization: frequency and duration of hospital admissions after HMV initiation, ICU admission and duration, respiratory and nonrespiratory physician out-patient clinic visits, same-day surgeries, emergency department visits, diagnostic tests including polysomnograms, home care services, and drug costs. We used validated methods for case-costing to determine health care costs in the year before the initiation of HMV.34 This case-costing approach does not include the cost of a ventilator because this information is not available in the health administrative databases.
Incidence
We defined incidence of HMV as the number of new HMV approvals in the ADP database per 100 000 children in Ontario per fiscal year. We used 2001 census data from the government of Canada for years 2003 to 2005, 2006 Census data for years 2006 to 2010, 2011 Census data for 2011 to 2015, and 2016 census data for 2016 to 2017.
Statistical Analyses
Patient characteristics and descriptive variables are presented for all children and subdivided by ventilation type. Baseline characteristics were compared between children receiving NIV and IMV using standardized differences. Crude incidence rates were reported per 100 000 children for each study year and by ventilation type. One-year mortality rates were calculated as the number of deaths per fiscal year divided by the number of new HMV approvals in the same year. These were then averaged over 3 time periods because of the small numbers that would potentially enable patient identification. We generated a Cox proportional hazards model of time to death after HMV initiation to investigate the effect of ventilation type (invasive or noninvasive) adjusting for the following covariates: (1) urban versus rural residence, (2) income quintile, (3) sex, (4) CCC category, (5) age at HMV initiation (0–5, 6–10, and 11–17 years), (6) number of medical technologies used at home, and (7) health care costs in the year before initiation of HMV. We used Statistical Analysis Systems 7.15 (SAS Institute, Inc., NC). All analyses were 2-tailed with significance set at P < .05.
Results
Cohort Characteristics
We identified 906 children approved for HMV over the 14 fiscal years. Most approvals (658, 73%) were for NIV, with 248 approvals (27%) for IMV. Children receiving IMV were younger (mean [SD], 6.0 [6.2] vs 9.2 [5.8]; standardized difference. 0.5) and used more medical technologies (mean [SD], 3.5 [1.9] vs 1.4 [1.3]; standardized difference, 1.3) with a smaller proportion not having a CCC (13 of 248 [5.2%] vs 125 of 658 [19%]; standardized difference, 0.4). Demographic characteristics are shown in Table 1.
Demographic Characteristics
. | Invasive Ventilation . | Noninvasive Ventilation . | Total . | Standardized Differencea . |
---|---|---|---|---|
Number of children | 248 | 658 | 906 | — |
Age, y | ||||
Mean (SD) | 6.0 (6.2) | 9.2 (5.8) | 8.4 (6.0) | 0.5 |
Median (IQR) | 4 (0–12) | 10 (4–14) | 9 (2–14) | — |
0–5 y | 137 (55.2) | 201 (30.5) | 338 (37.3) | 0.5 |
6–10 y | 34 (13.7) | 131 (19.9) | 165 (18.2) | 0.2 |
11–17 y | 77 (31) | 326 (49.5) | 403 (44.5) | 0.4 |
Sex (males) | 152 (61.3) | 409 (62.2) | 561 (61.9) | 0.02 |
Neighborhood income quintile, mean (SD) | ||||
Quintile 1 (lowest) | 65 (26.2) | 154 (23.4) | 219 (24.2) | 0.06 |
Quintile 2 | 43 (17.3) | 126 (19.1) | 169 (18.7) | 0.05 |
Quintile 3 | 42 (16.9) | 128 (19.5) | 170 (18.8) | 0.07 |
Quintile 4 | 56 (22.6) | 121 (18.4) | 177 (19.5) | 0.1 |
Quintile 5 | 41 (16.5) | 125 (19) | 166 (18.3) | 0.07 |
Rural residence | 25 (10.1) | 92 (14) | 117 (12.9) | 0.1 |
Neuromuscular condition | ||||
Muscular dystrophy | 50 | 127 | 177 | 0.02 |
Myasthenia gravis | 6 | 0 | 6 | — |
Multiple sclerosis | 7 | 7 | 14 | 0.1 |
Cerebral palsy | 29 | 54 | 83 | 0.1 |
Spinal muscular atrophy | 17 | 40 | 57 | 0.03 |
Nonspecific neuromuscular disorder | 15 | 11 | 26 | 0.2 |
Other conditions | ||||
Diaphragm paralysis | 14 | 11 | 25 | 0.2 |
Kyphoscoliosis | 47 | 111 | 158 | 0.05 |
Obesity | 16 | 93 | 109 | 0.3 |
Spina bifida | 9 | 23 | 32 | 0.01 |
Pulmonary congestion | 19 | 30 | 49 | 0.1 |
Asthma | 82 | 174 | 256 | 0.2 |
Sepsis | 152 | 206 | 358 | 0.6 |
Pneumonia | 104 | 178 | 282 | 0.3 |
CCC category | ||||
No CCC | 13 | 125 | 138 | 0.4 |
Neurologic impairment only | 14 | 92 | 106 | 0.3 |
Single-organ CCC | 52 | 199 | 251 | 0.2 |
Multiorgan CCC | 169 | 242 | 411 | 0.7 |
Number of technologies used at home | ||||
Mean (SD) | 3.5 (1.9) | 1.4 (1.3) | 1.9 (1.8) | 1.3 |
Median (IQR) | 3 (2–5) | 1 (0–2) | 2 (0–3) | — |
. | Invasive Ventilation . | Noninvasive Ventilation . | Total . | Standardized Differencea . |
---|---|---|---|---|
Number of children | 248 | 658 | 906 | — |
Age, y | ||||
Mean (SD) | 6.0 (6.2) | 9.2 (5.8) | 8.4 (6.0) | 0.5 |
Median (IQR) | 4 (0–12) | 10 (4–14) | 9 (2–14) | — |
0–5 y | 137 (55.2) | 201 (30.5) | 338 (37.3) | 0.5 |
6–10 y | 34 (13.7) | 131 (19.9) | 165 (18.2) | 0.2 |
11–17 y | 77 (31) | 326 (49.5) | 403 (44.5) | 0.4 |
Sex (males) | 152 (61.3) | 409 (62.2) | 561 (61.9) | 0.02 |
Neighborhood income quintile, mean (SD) | ||||
Quintile 1 (lowest) | 65 (26.2) | 154 (23.4) | 219 (24.2) | 0.06 |
Quintile 2 | 43 (17.3) | 126 (19.1) | 169 (18.7) | 0.05 |
Quintile 3 | 42 (16.9) | 128 (19.5) | 170 (18.8) | 0.07 |
Quintile 4 | 56 (22.6) | 121 (18.4) | 177 (19.5) | 0.1 |
Quintile 5 | 41 (16.5) | 125 (19) | 166 (18.3) | 0.07 |
Rural residence | 25 (10.1) | 92 (14) | 117 (12.9) | 0.1 |
Neuromuscular condition | ||||
Muscular dystrophy | 50 | 127 | 177 | 0.02 |
Myasthenia gravis | 6 | 0 | 6 | — |
Multiple sclerosis | 7 | 7 | 14 | 0.1 |
Cerebral palsy | 29 | 54 | 83 | 0.1 |
Spinal muscular atrophy | 17 | 40 | 57 | 0.03 |
Nonspecific neuromuscular disorder | 15 | 11 | 26 | 0.2 |
Other conditions | ||||
Diaphragm paralysis | 14 | 11 | 25 | 0.2 |
Kyphoscoliosis | 47 | 111 | 158 | 0.05 |
Obesity | 16 | 93 | 109 | 0.3 |
Spina bifida | 9 | 23 | 32 | 0.01 |
Pulmonary congestion | 19 | 30 | 49 | 0.1 |
Asthma | 82 | 174 | 256 | 0.2 |
Sepsis | 152 | 206 | 358 | 0.6 |
Pneumonia | 104 | 178 | 282 | 0.3 |
CCC category | ||||
No CCC | 13 | 125 | 138 | 0.4 |
Neurologic impairment only | 14 | 92 | 106 | 0.3 |
Single-organ CCC | 52 | 199 | 251 | 0.2 |
Multiorgan CCC | 169 | 242 | 411 | 0.7 |
Number of technologies used at home | ||||
Mean (SD) | 3.5 (1.9) | 1.4 (1.3) | 1.9 (1.8) | 1.3 |
Median (IQR) | 3 (2–5) | 1 (0–2) | 2 (0–3) | — |
IQR, interquartile range. —, values not calculated.
Standardized difference between invasive and noninvasive ventilation. Standardized difference <0.1 indicates the difference between 2 groups is negligible.
HMV Incidence
The mean (SD) crude incidence rate over the 14-year period was 2.4 (0.6) per 100 000 children. The crude incidence rate by fiscal year for all HMV approvals increased from 2.1 per 100 000 children (2003) to 2.9 per 100 000 children (2016) (Fig 1, Supplemental Table 4). This represents a 37% increase. The crude incidence rate for IMV approvals between 2003 and 2016 increased by 34% from 0.6 to 0.8 per 100 000 children and NIV by 37% from 1.5 to 2.1 per 100 000 children.
Crude incidence rates per 100 000 of approved ADP ventilator applications for children from 2003 to 2016 in Ontario, Canada.
Crude incidence rates per 100 000 of approved ADP ventilator applications for children from 2003 to 2016 in Ontario, Canada.
Mortality
Mortality rates over the 14-year period were as follows: 2003 to 2007, 5.0%; 2008 to 2012, 4.4%; and 2013 to 2016, 4.5% (Table 2). However, these rates fluctuated throughout the study period with no consistent decreasing trend.
One-Year Mortality Rates After Pediatric Home Mechanical Ventilation Initiation Between 2003 and 2016 in Ontario, Canada, Averaged Over 3 Time Periods
Year Grouping . | Mortality Rate (%) . |
---|---|
2003–2007 | 5.0 |
2008–2012 | 4.4 |
2013–2016 | 4.5 |
Year Grouping . | Mortality Rate (%) . |
---|---|
2003–2007 | 5.0 |
2008–2012 | 4.4 |
2013–2016 | 4.5 |
Mortality rates are presented as averages over 3 time periods instead of yearly because of the small numbers of the annual mortality rate that could allow for patient identification.
Compared with those children who were invasively ventilated, we found increased mortality was associated with noninvasive ventilation, (adjusted hazard ratio [aHR], 1.9; 95% confidence interval [CI], 1.3–2.8), children from families in the lowest income quintile compared with the highest quintile, (aHR, 2.5; 95% CI, 1.5–4.0), having neurologic impairment CCC (as compared with no CCC) (aHR, 2.9; 95% CI, 1.4–6.4), aged 11 to 17 years (compared with 0–5 years) at time of ventilation initiation (aHR, 1.5; 95% CI, 1.1–2.0), and those children having higher health care costs in the 1 year before HMV initiation (aHR, 1.5; 95% CI, 1.3–1.7) (Table 3).
Summary of Cox Proportional Hazards Model Of Survival Analysis Based on Sociodemographic Factors
Variable . | Unadjusted HR . | aHR . | ||
---|---|---|---|---|
HR . | 95% CI . | HR . | 95% CI . | |
Type of HMV | ||||
Noninvasive ventilation | 1.0 | 0.8–1.4 | 1.9a | 1.3–2.8 |
IMV (ref) | — | — | — | — |
Area of residence | ||||
Urban | 1.1 | 0.7–1.8 | 1.0 | 0.7–1.5 |
Rural | — | — | — | — |
Neighborhood income quintile | ||||
Quintile 1 (lowest) | 2.3 | 1.4–3.7 | 2.5a | 1.5–4.0 |
Quintile 2 | 1.4 | 0.8–2.4 | 1.5 | 0.9–2.6 |
Quintile 3 | 1.9 | 1.2–3.2 | 2.1a | 1.3–3.5 |
Quintile 4 | 1.6 | 1.0–2.7 | 1.9a | 1.1–3.2 |
Quintile 5 (ref) | — | — | — | — |
Sex | ||||
Male | 1.0 | 0.8–1.4 | 1.2 | 0.9–1.5 |
Female | — | — | — | — |
CCC category | ||||
Neurologic impairment only | 4.9 | 2.3–10.3 | 2.9a | 1.4–6.4 |
Single-organ system | 3.3 | 1.6–6.6 | 1.9 | 0.9–4.1 |
Multiple-organ system | 4.4 | 2.2–8.6 | 2.0 | 0.9–4.2 |
No CCC (ref) | — | — | — | — |
Age, y | ||||
6–10 | 0.4 | 0.3–0.7 | 0.7 | 0.4–1.2 |
11–17 | 0.9 | 0.7–1.2 | 1.5a | 1.1–2.0 |
0–5 (ref) | — | — | — | — |
Number of medical technologies | 1.2 | 1.1–1.4 | 1.0 | 0.9–1.2 |
Health care costs 1 y before HMV initiation | 1.3 | 1.3–1.5 | 1.5a | 1.3–1.7 |
Variable . | Unadjusted HR . | aHR . | ||
---|---|---|---|---|
HR . | 95% CI . | HR . | 95% CI . | |
Type of HMV | ||||
Noninvasive ventilation | 1.0 | 0.8–1.4 | 1.9a | 1.3–2.8 |
IMV (ref) | — | — | — | — |
Area of residence | ||||
Urban | 1.1 | 0.7–1.8 | 1.0 | 0.7–1.5 |
Rural | — | — | — | — |
Neighborhood income quintile | ||||
Quintile 1 (lowest) | 2.3 | 1.4–3.7 | 2.5a | 1.5–4.0 |
Quintile 2 | 1.4 | 0.8–2.4 | 1.5 | 0.9–2.6 |
Quintile 3 | 1.9 | 1.2–3.2 | 2.1a | 1.3–3.5 |
Quintile 4 | 1.6 | 1.0–2.7 | 1.9a | 1.1–3.2 |
Quintile 5 (ref) | — | — | — | — |
Sex | ||||
Male | 1.0 | 0.8–1.4 | 1.2 | 0.9–1.5 |
Female | — | — | — | — |
CCC category | ||||
Neurologic impairment only | 4.9 | 2.3–10.3 | 2.9a | 1.4–6.4 |
Single-organ system | 3.3 | 1.6–6.6 | 1.9 | 0.9–4.1 |
Multiple-organ system | 4.4 | 2.2–8.6 | 2.0 | 0.9–4.2 |
No CCC (ref) | — | — | — | — |
Age, y | ||||
6–10 | 0.4 | 0.3–0.7 | 0.7 | 0.4–1.2 |
11–17 | 0.9 | 0.7–1.2 | 1.5a | 1.1–2.0 |
0–5 (ref) | — | — | — | — |
Number of medical technologies | 1.2 | 1.1–1.4 | 1.0 | 0.9–1.2 |
Health care costs 1 y before HMV initiation | 1.3 | 1.3–1.5 | 1.5a | 1.3–1.7 |
Ref, reference. —, values not calculated.
Statistically significant.
Discussion
In this population-based, 14-year cohort of children receiving HMV in Ontario Canada, we found that the incidence of HMV use increased. The following essentially nonmodifiable factors were associated with increased mortality: NIV, low neighborhood income, CCC because of neurologic impairment, older-aged children, and higher health care costs in the year before ventilation initiation.
The incidence rate of pediatric HMV in Ontario increased primarily because of increased use of NIV. Our results are in keeping with an older study from British Columbia, Canada, that reported a significant rise in the incidence rate of pediatric HMV from 1 child per million in 1996 to 14 children per million in 2006 that was also linked to a significant increase in use of NIV.2 NIV is now a widely accepted treatment option for children with sleep-disordered breathing and neuromuscular disease, 2 common indications for pediatric HMV.2,7
We report higher mortality in children using NIV compared with IMV, which is contrary to the findings of previous studies. A prospective study in British Columbia of 144 children over a 15-year period by McDougall et al reported 10-year survival rates of 97% and 77% for NIV and IMV, respectively.2 Children on NIV may be just as medically fragile as children receiving IMV. In fact, some of these families may have declined a tracheostomy because of its invasiveness and/or incongruence with their goals of care. This may be 1 of the reasons for the fall in pediatric tracheostomy rates in the United States from mean (SD) 6.8 (0.2) in 2000 to 6.0 (0.2) in 2012 using a national database of hospital discharge data.35 Children receiving IMV in Ontario have some guaranteed home nursing support because of necessary tracheostomy care, whereas this is often not the case for children receiving NIV.
In our cohort, mortality rates remained consistent throughout the 14-year study period. We elected to report mortality rates as the number of deaths per year divided by new HMV approvals to reduce the risk of survivor bias. Our findings highlight that the risk of death in a child receiving HMV remains an important consideration for families and care providers. Risks related to the use of mechanical ventilation at home that can lead to mortality include ventilator/equipment malfunction, aspiration, infection, tracheostomy and circuit obstruction, and accidental tracheostomy tube decannulation.36 Additionally, progression of the child’s underlying medical disorder may result in death.
Providing care for a child who is medically complex and dependent on medical technology is expensive. Although children receiving long-term ventilation at home continue to have hospital admissions after HMV initiation, days in hospital significantly decrease. In a retrospective cohort of children receiving HMV via tracheostomy, the initial hospital admission when HMV was initiated was a mean of 155 days compared with a mean of 58 days in hospital for subsequent admissions.37 However, HMV shifts the burden of costs and responsibility to the child’s family.19 Notably, we demonstrated that increased mortality was associated with low annual neighborhood income. Out-of-pocket expenses include transportation costs for clinic visits, mobility devices (eg, wheelchairs), and additional medications, supplies, equipment, and home nursing supports not covered by government assistance programs or insurance providers.4,14,38,39 Seear et al recruited 90 HMV children in British Columbia and found that additional self-funded nursing supports could cost families up to $3500 Canadian dollars per month, medications up to $850 per month, and out- of-pocket supplies up to $2000 per month.40 Families in lower income strata may not be able to afford this level of expense, which may account for the association we found between lower neighborhood income and increased mortality. Furthermore, family caregivers experience loss of income, causing further financial strain. Clinical guidelines from the American and Canadian Thoracic Societies mandate “24/7 eyes on care” by an awake and alert caregiver for all HMV children because short delays in recognition and response can have highly deleterious consequences.38,41 As a result, 1 parent often remains at home to be the primary caregiver. Thus, shared decision-making among children, their families, and care providers regarding HMV initiation should include a clear discussion regarding the financial implications of supporting a child on HMV.
We found that children who were older at the time of HMV initiation and those diagnosed with a neurologic CCC had increased mortality compared with younger children and those with other types of CCC. This may be because of older children having further disease progression, such as those with spinal muscular atrophy or Duchenne muscular dystrophy.14,16,23,26 Previous studies report up to 85% of deaths in children receiving HMV result from disease progression.
Limitations
Our study has some notable limitations. First, our findings may not be generalizable to children and families in other Canadian provinces or other jurisdictions where different HMV prescribing patterns may exist. Second, as a retrospective cohort study, our results may be confounded by factors not captured in health administrative databases. Third, we are unable to determine causation and can only suggest associations between the variables studied and their potential impacts on mortality.
Conclusions
The incidence of pediatric HMV is increasing in Ontario, primarily because of increased prescription of NIV. Mortality rates are higher in children receiving NIV, those in families with low income, those with neurologic impairment, those of older age, and those having higher health care costs in the year before ventilation initiation. Although many of these factors are largely nonmodifiable, clinicians should consider financial costs and expenses to families when designing pediatric HMV programs and advocate for increased financial support for lower income families. Future research is needed to investigate the effect of targeted financial supports for those HMV children at increased risk of mortality.
Parts of this material are based on data and information compiled and provided by the Canadian Ministry of Health and the Canadian Institute for Health Information. Geographical data are adapted from Statistics Canada, PCCF+ version 6D, 2009 to 2013. This does not constitute endorsement by Statistics Canada of this product. The analyses, conclusions, opinions, and statements expressed herein are solely those of the authors and do not reflect those of the funding or data sources; no endorsement is intended or should be inferred.
The data set from this study is held securely in coded form at ICES. Although legal data sharing agreements between ICES and data providers (eg, health care organizations and the government) prohibit ICES from making the data set publicly available, access may be granted to those who meet prespecified criteria for confidential access, available at www.ices.on.ca/DAS (e-mail: [email protected]). The full data set creation plan and underlying analytic code are available from the authors upon request, understanding that the computer programs may rely upon coding templates or macros that are unique to ICES and are therefore either inaccessible or may require modification.
Drs Amin and Rose conceptualized the study, acquired the funding, analyzed and interpreted the data, and drafted the initial manuscript and subsequent revisions; Dr Verma analyzed and interpreted the data, and drafted the initial manuscript and subsequent revisions; Mr Qing Bai analyzed and interpreted the data, and drafted the initial manuscript; Drs Cohen, Guttmann, Gershon, Katz, and Lim interpreted the data, and drafted the initial manuscript and subsequent revisions; and all authors approved the final manuscript as submitted and agree to be accountable for all aspects of the work.
FUNDING: Supported by ICES, which is funded by an annual grant from the Ontario Ministry of Health and the Ministry of Long-Term Care. This study also received funding from the Ontario Thoracic Society.
CONFLICT OF INTEREST DISCLOSURE: The authors have indicated they have no conflicts of interest relevant to this article to disclose.
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