Discussion
Using a nationwide claims database, this is one of the first detailed epidemiological studies on children receiving HHD, providing fundamental information. The study revealed a wide variation in when and how many children could be released from their HHD and identified risk factors for increased HHD use during the 5 year follow-up period. The provided information could guide healthcare professionals, improving the quality of life of children receiving HHD and their families and facilitating health policymakers to consider measures.
The number of children receiving HHD increased each year (figure 2), while the number of newborn infants decreased,17 although several factors may have affected the number of children receiving HHD, such as the revision of the fee table, practice in coding/claiming and changes in home healthcare delivery system. This is partly because of the increase in advanced maternal age following women’s empowerment, fertility treatments and advancements in home medical care (deinstitutionalisation). Additionally, the cohort gradually became severe/complex, owing to the increase in the proportion of devices, which required more resources such as mechanical ventilation and multiple devices. These results imply that the burden on children receiving HHD and healthcare providers will continue to increase. Family caregivers highlighted the financial burden associated with the care of a child requiring medical technology,18 while financial difficulties could be universal for all families of children with chronic illness.19 20 This study reconfirms the need for evidence-based, systematic, comprehensive and immediate action to address this situation.
From the perspective of the types of children receiving HHD, the results indicated a stable decrease in the number of children receiving HHD using tracheostomy care, partly owing to the dissemination of noninvasive respiratory home care, such as NPPV. Some families may have rejected tracheostomy because of its invasiveness and incongruence with their care goals. An increase in noninvasive respiratory home care has also been reported internationally.21
In terms of international comparison, the male-to-female ratio (53.7:46.3) was comparable with previous similar studies from the USA, where children used home healthcare in the Medicaid cohort (54.5:45.5)3 and Canada, where children in Ontario with medical complexity had a ratio of 55.4:44.6.2 From the available data, we found that (1) the crude incidence rate of children receiving home mechanical ventilation in Japan (4.1 per 100 000 children; online supplemental eTable 1) was higher than that in Canada (2.9 per 100 000 children) and (2) there was an increasing trend of these children in both Canada and Japan.22 Interpreting this comparison is challenging, as differences in morbidity, applicable criteria of HHD, intensity of care (withdrawal or not), family involvement in medical decision-making, healthcare system and culture may contribute to these variations. While direct comparison is difficult owing to the differences in ways to identify children receiving HHD, its definition, the terminology used to describe and classify CMC23 and the structure of the database, further international comparison is needed to facilitate a better understanding of the epidemiology of children receiving HHD and improve their quality of life.
Although the data were not risk-adjusted, there was wide variation in when patients could (not) be released from their HHD (figure 3). To our knowledge, this is one of the first reports on the timing of HHD worldwide. While guidelines for discontinuing home oxygen therapy in children are available,24 few recommendations exist for other HHD. Therefore, the weaning process of mechanical ventilation is based on clinical practice,25 and there is no guidance on the timing of tracheostomy closure in children.26 Further empirical data, evidence and recommendations would help understand and improve their health conditions and quality of life, including that of their families. Especially, further research on differences in morbidity, applicable criteria of HHD, intensity of care (withdrawal or not), family involvement in medical decision-making and healthcare system, which may contribute to variations in the timing of HHD release, is strongly required.
We found that neurological diseases were risk factors for increased home device use, confirming expectations but providing a first-time assessment of their impact. These neurological diseases, including intractable ones, generally have a poor prognosis and seem to contribute to this association. Additionally, we observed that low birth weight was associated with a higher risk of increased use of home devices, whereas very low birth weight was not. We suspect that this is partly because low-birth-weight infants were likely to have other comorbidities related to neurological diseases or several more severe comorbidities than very-low-birth-weight infants, and very low birth weight infants tended to be excluded from the cohort (owing to death during the 5 year follow-up and longer hospitalisation as they could not be discharged home). Further investigation into detailed major diagnoses is worthwhile to understand the characteristics of children receiving HHD for each disease.
Regarding regional variations in following up children receiving HHD, there was wide variation in the types of institutions (online supplemental eFigure 1). A similar regional variation was reported in the USA regarding the types of care and EDs.27 28 We suspect that this septal variation was related to available healthcare/community resources by region (which was not included in our dataset), clinical practice for severely ill children, including withholding/withdrawal of intensive treatment and recognition of best primary care (family-centred medical home or not).29 Indeed, a previous study indicated numerous gaps in the current system for transitioning CMC from hospital to home healthcare.30
Paediatricians and healthcare providers must understand the complexities of a child’s medical needs, family needs, prognosis, end-of-life care and available community resources.31 However, Foster et al reported that home healthcare for CMC is a ‘patchwork of policies and programmes’ that does not align with the needs of many children; there seems to be a lack of policy programmes for these children.32 Further research is required to improve policymaking regarding children receiving HHD.
Strengths and limitations
This study used a nationwide administrative database that included most children receiving HHD and had the strengths of large sample size and representativeness. Furthermore, the database enabled nationwide follow-up of the study cohort. However, this study has certain limitations. First, confounding factors could not be controlled for. For example, there is a lack of clinical information regarding the severity of each disease, despite its wide variation. Second, our study followed the children for only 5 years, and we did not follow up children receiving HHD aged ≥19 years. Third, there is the possibility of underestimation and overestimation. The number of children receiving HHD for each type of device was sometimes underestimated and/or overestimated, partly owing to the revision of the fee table every 2 years. Major changes are as follows: (1) we showed two types of tube feeding owing to the changes in the fee table in 2012 and (2) we revised the definition of home oxygen (deregulation of applying the fee in 2016). In general, the revision enables more precise capture of children receiving HHD. The number of children receiving HHD could also be overestimated because we could not detect all deaths. Deaths not resulting in an insurance claim at the time of occurrence, such as accidental deaths and those discovered later, could not be identified. The duration of HHD might be underestimated or overestimated as no precise algorithm has been established to detect the continuous use of HHD. Fourth, we did not include the socioeconomic context of children receiving HHD or families, which is currently reported to be associated with adherence to patient-centred medical home visits.33 Finally, there may be differences in the criteria for applying HHD (discharge to home or hospitalisation) among institutions/regions. We did not investigate the supply and demand of home devices for needy children. We also did not consider therapy compliance due to data unavailability, which may affect health resource use and outcome.