This study is the first to provide an estimate of number of children and young adults living with life-limiting conditions and receiving specialized palliative care for the world’s largest ethnic group, Han Chinese. The overall prevalence of life-limiting conditions in Taiwan has increased markedly over this ten-year study period, from 59.4 per 10,000 population (95% CI: 58.8–59.9) in 2008 to 81.0 per 10,000 population (95% CI: 80.3–81.7) in 2017. Comparable to previous studies, the prevalence of life-limiting conditions was higher in boys than girls [1, 7]. However, it was observed that girls were more prone to clinical instability than boys [2], such as having a higher risk for urinary tract infection complications [22]. The prevalence of life-limiting conditions in Taiwan was over two times greater than previous estimation for Chinese population in England in 2017 [1]. Different from previous studies [1, 7], the prevalence of life-limiting conditions was highest for oncological disorders in Taiwan, but the annual trend remained relatively static. The exclusion of diagnoses through ICD-10 might lead to a reduction in the reported prevalence of oncological diseases between 2016 and 2017. However, the prevalence of oncology diseases (avg. 21.8 per 10,000 over the ten year) was still higher than that reported in England [1] and other countries with high social-demographic indexes (16.2 per 10,000) [23]. Previous study showed a higher rate of childhood hepatoblastoma in Taiwan (2.67 per million person-y) than the rates in Europe (1.5 per million person-y) and the USA (2.2 per million person-y), which may be related to the genetic background of the Han Chinese ethnicity [24]. As the NHI and the Childhood Cancer Foundation of Republic of China has covered the expenses of cancer treatment, as well as the case management system is increasingly being developed in hospitals, treatment compliance and survival rates for children and young adults with cancer have improved [25, 26]. For instance, the 5-year overall survival rates for children with acute myeloid leukemia without relapse significantly increased from 50.8% during 1996–2007 to 72.5% during 2008–2019 in Taiwan. This may be attributed to the improvement of access to treatments and supportive care significantly decreased death caused by infection [26]. Our study also found a steep decline in use of traditional Chinese medicine, which could be attributed to improved treatment adherence and increased awareness of cancer [27].

Our research has revealed a significant rising trend in the prevalence of congenital anomalies (p < .001), making it the most prevalent group since 2017 (20.8 per 10,000 population). This rise may be partly due to the change from using the list of converted ICD-9 codes to the list of diagnostic ICD-10 codes, resulting in an underestimated number of recorded diagnoses between 2008 and 2015. Despite the incidence of newborns with congenital anomalies showing a significant downward trend between 2004 and 2011 [28], congenital malformations remain the leading cause of infant mortality in Taiwan [29]. A previous study reported that less than 30% of Taiwanese children and teenagers with congenital heart disease complied with continual cardiac follow-up care up to their teenage years, potentially leading to avoidable complications and subsequent mortality [30]. With advances in diagnosis, medical technology, surgical interventions, and integrative transitional care, children and young adults were able to live through adulthood [30, 31]. It was predicted that congenital disorders will still be the largest group of life-limiting conditions and will rise to a prevalence of 37.2 per 10,000 population by 2030 [1].

Similarly, as Taiwan’s NHI approved gene therapy for spinal muscular atrophy in children in 2023 [32], future trends to show a rise in the prevalence of neurological diseases may be expected due to an increase in survival rates. During the years of this study, there has been an increase in the prevalence of hematological diseases, which could be attributed to the promotion of multiple screening policies. For instance, Taiwan began a national strategy in 2005 for the elimination of mother-to-child transmission of HIV by offering free testing at antenatal care and medical care services for pregnant women and suspected HIV-infected infants [33]. Since 2017, comprehensive prevention programs and treatment strategies have led to a decrease in HIV transmission and a reduction in the HIV epidemic in Taiwan [34]. With a reduced likelihood of late HIV diagnosis and HIV-related mortality, including suicide and accident mortality [35], this prevalence trend is expected to continue increasing.

Our study found a similar prevalence of genitourinary diseases as compared with that in England [1] and a significant rising trend (p = .001). Even though Asian children and adolescents have been reported to have the lowest incidence and prevalence of end-stage renal disease, Taiwan has reported the highest prevalence of individuals with end-stage renal disease undergoing maintenance dialysis globally [36]. Asians having been found to have higher preferences for life-sustaining treatments to prolong life may thus be one the reasons to have a higher prevalence than Western populations [1, 7]. Expanding specialized palliative care service coverage in Taiwan since 2009 led to less use of life-sustaining treatments for elderly patients with end-stage renal disease [37], but the impact on the pediatric population is unclear.

We found the gap between the number of children and young adults with life-limiting conditions and the actual annual number of people receiving specialized palliative care and other healthcare services. Although there was a decline in the annual death rates among children and young adults with life-limiting conditions, which was 21.5 per 1,000 in 2017, it was much lower than the rate of receiving specialized palliative care services. The proportion of patients and families needing specialized palliative care will vary from place to place, depending on the skills and resources available through primary care [38]. Previous studies interviewing Taiwanese pediatric healthcare professionals suggest that refractory pain may be better managed if the child was cared for in an inpatient hospice than by a pediatric primary care team [39]. Although we did find that specialized palliative care services, including shared care, palliative physician and nurse visit, have increased in use over time in Taiwan, inpatient hospice care has a slight decrease since 2012. All children and young adults who receive palliative care services were coded, so it is unlikely that the low percentage of patients is due to a lack of coding for services. For some Taiwanese parents, transferring their child to an inpatient hospice indicated they had given up on their child [11, 40]. A previous study found that two-thirds of Taiwanese pediatric patients who died of cancer received ICU care in their last month of life. This proportion was higher than what was reported in the United States and Korea [41]. Therefore, instead of transferring to inpatient hospices, which were located in an adult care setting, Taiwanese children and young adults may prefer a shared care model where they could be cared for by their primary pediatric care team. This also indicated an increasing need for adequate transition [31] and advance care planning for adolescents and young adults [42], which both currently lack service access in Taiwan.

This study also found that people using general home care service was over 60 times higher rates than those receiving specialized palliative home visits in 2017. One possible reason for this was the fact that specialized palliative care services can only be covered by the NHI if the patients were in at least one of the following life-limiting conditions: advanced cancer, terminal motor neuron disease, senile and presenile organic psychosis, cerebral degeneration, heart failure, chronic airway obstruction, other lung diseases, chronic liver disease and cirrhosis, and renal failure [18]. These conditions are generally adult specific and not relevant to children. After comparing ICD codes of the above diseases [43] with the coding framework of life-limiting conditions [1, 7], we found none of the ICD codes in the hematological, metabolic, circulatory, congenital abnormalities, perinatal, and “other” groups were eligible for specialized palliative care services coverage by Taiwan’s NHI. Therefore, we recommend that policies are needed for expansion of specialized palliative care services to ensure that service access is available to children and young adults in all diagnostic groups.

Despite the observed low rate of palliative home care use, one should be cautious to conclude that children and young adults prefer hospital-based services (shared care or inpatient hospice) over community-based care options [44]. Previous studies suggested that among Taiwanese children and young adults with cancer, specialized palliative care team were involved at a later stage of the disease, with more than one-fifth of them receiving such care within their last 3 days of life [45]. This usually results in not enough time for setting up the outpatient setting, such as adequate equipment available in the home or an access to a palliative care specialized contact. In Taiwan, only persons with specialized training in palliative care can deliver palliative home care, but they may lack pediatric palliative care training. The first documented professional training for pediatric palliative care began in 2014 by the Taiwan Association of Hospice Palliative Nursing, but only included two hours of lecturing in the total 15 days of palliative care training and the practicum [46]. A lack of trust in an adult patient-oriented specialized palliative care team may be one of the reasons for the low rate of use in children and young adults.

This study also found an increased usage of chronic care for ventilator-dependent patients (p < .001). This may indicate a growing number of families were home-caring for children and young adults who were dependent on ventilators and other highly technical care. For patients with long-term care needs, with the family having ability to care independently for their child, some suggested that palliative home care provides an important option for respite care [40]. To improve community-based palliative care for this population, policy within the NHI in Taiwan should emphasize home-based multi-disciplinary approaches to support family caregivers, e.g. in-reach emergent technical support or short breaks provided at home. Coordinated multidisciplinary and multiagency support should be readily available, with clear information provision and easy routes of access [47].

Traditional Chinese medicine covered by Taiwan’s NHI includes powdered Chinese herbal preparations, acupuncture, electropuncture, moxibustion, and manipulative therapy, health and nutrition education, and is highly used among children and young adults with life-limiting conditions. It has been previously found that children and adolescents with cancer, who suffered from drowsiness, lack of energy, and pain were more likely to use acupuncture [48]. Although there is good evidence in the literature that traditional Chinese medicine helps reduce cancer-related fatigue and chemotherapy-induced nausea and vomiting, and lowering infection rates in patients with leukemia [49], there is conflicting evidence regarding the treatment for dyspnea [50]. More work is needed to understand the effectiveness of traditional Chinese medicine in palliative care.

Previous study estimates 42% parents reported making use of psychological support services [51], which usually included self-support groups, psychological, and spiritual counselling [52]. Our findings expand the knowledge in the pediatric literature that there is a growing trend in the patient use of psychotherapy. Mental health professionals should be prepared to care for a growing number of children and young adults with life-limiting conditions with basic knowledge of palliative care. They should also be included within pediatric palliative care networks to ensure good co-ordination of care.

This is the first population-based study investigating specialized palliative care utilization in children and young adults with a life-limiting condition. The data sources were from national datasets; therefore, there was a strong backup for uploaded demographic information and reliable service use information. This study differs from a previous one [1] in that it only includes individuals who have had an inpatient or outpatient service recorded for a life-limiting condition in the current year. This ensures that the individuals being considered are still in need of medical care and are currently experiencing life-limiting conditions. While this may result in a decrease in estimated prevalence due to patients lost to follow-up [30], it ultimately increases accuracy.

This study has limitations. Firstly, there may have been lost cases from those who survived after a perinatal life-limiting condition and did not have a record of life-limiting condition diagnosis after their NHI enrollment procedures were completed (which should be done within 60 days of birth). However, this also means that we may have less numbers of infants who had a life-threatening event around the time of birth but were no longer considered as having a life-limiting condition in the year of birth, compared with the previous study [1].

Secondly, about 10% of life-limiting condition ICD-10-CM codes did not have a corresponding ICD-9-CM code; therefore, we may have underestimated the prevalence of life-limiting conditions between 2008 and 2015. Thirdly, a child or young adult was only required to have one recording of a life-limiting condition in any clinic or hospital to be included in these analyses. Children and young adults diagnosed with cancer may remain this record even if they had achieved full remission and were no longer life-limiting. Therefore, we may be capturing some cancer survivors who may not require palliative care. If individuals were not admitted to a clinic or hospital in a particular year, they were excluded from the prevalence estimation. As a result, individuals with poor compliance to medical follow-ups may be missed. Moreover, the diagnosis’s staging and the disease’s severity were unknown due to data unavailability. It is unclear whether individuals in each diagnostic group with specialized palliative care received timely access. Finally, this study did not include those with uninsured healthcare services, e.g., pastoral care and bereavement services, which were highly been used in parents of neonates [51].