We designed a quasi-experimental pre-post intervention study in the Complex Chronic Patient and Palliative Care Unit of a pediatric tertiary hospital.

The study population comprised children with CCCs or those needing palliative care and their families. Inclusion criteria required that children meet the definition of medical complexity algorithm defined by the Seattle Children’s Hospital Group and score at least 6,5 points on the PedCom Scale [25,26]. Participants also had to be followed-up in the Complex Chronic Patient and Pediatric Palliative Care Unit of the hospital and had a high level of complexity, defined not only by the identification of the children as complex chronic patients but also by high consumption of resources: two or more hospital admissions, at least one admission to intensive care, or six or more visits to the emergency department in the last twelve months [27]. Patients older than 18 years or those whose families did not provide informed consent were excluded.

The study protocol was in accordance with the Declaration of Helsinki. All caregivers were informed about the study and signed a consent form, with no financial compensation provided. All data were anonymized to ensure confidentiality. This study was approved by the Research Ethics Committee of Malaga in May 2017 (reference: PIN-0287‐2016).

The telemedicine program was remotely managed and controlled by the hospital and its technical support center. It comprised three main functionalities:

During recruitment, to avoid selection bias caused by the digital divide for families who did not have internet access at home, it was provided through project funding.

The evaluation of the program was undertaken through the comparison of two one-year time periods; before the pandemic (March 2019 to February 2020) and during the pandemic (March 2020 to February 2021).

Sociodemographic variables described included age, gender, primary caregiver (parent or both parents), number of siblings, place of residence, and country of origin.

The evaluation of the telemedicine program was carried out with periodic assessments (ie, baseline, 3, 6, and 12 months) of the health care variables and caregiver reported outcomes. The last assessment was carried out 12 months after the start of the intervention. The health care data was collected by telephone and the tests by mail and email.

Data analysis was performed using free R software (version 4.0.2; R Foundation for Statistical Computing). The Shapiro-Wilk test analysis was performed to determine the normality of the study variables. Data were presented as mean (SD) values in normal distributions or as median (IQR) for non-normal distributions. For independent quantitative variables, Student one-tailed t test was used for normally distributed data, and the Mann-Whitney U test for not normal distributed data. The Wilcoxon signed-rank test was performed to analyze differences in the non-normal distributions, and the paired t test for normal distributed data. To compare qualitative variables, the χ² test was used for independent samples and McNemar test for related samples. A P value <.05 was considered statistically significant. P values were adjusted using the Benjamini-Hochberg correction to control for multiple comparisons.

A total of 34 patients were included, ranging in age from 3 months to 14.2 years, with a median age of 4.9 years; 53% (18/34) were female participants. The most common CCC that affected our patients was neurological 73% (n=25), followed by gastrointestinal 71% (n=24), respiratory 50% (n=17), cardiovascular 41% (n=14), and oncological 21% (n=7). In addition, neurological CCCs were also the most frequent primary CCC in 47% (n=16), with cerebral palsy being the most prevalent neurological disease (11/16, 69%).

The primary caregiver was the mother in 82% (n=28) and 47% (n=16) of the families received social assistance; 91% of the families (n=31) received psychological care. A total of 29% (n=10) of the families were from a foreign country and 41% (n=14) had no other children; the number of children per family ranged from 0 to 5, with a median of 1.

Regarding scheduled care, bivariate analysis showed that the mean number of visits to primary care consultations per year was higher in the pre-pandemic period (P=.03), as well as more hospital specialty consultations (P=.03) (Table 1).

In terms of emergency care, there were significant differences in the number of emergency department visits per year during the compared to the prepandemic period (P=.01), with fewer visits during the pandemic. No significant differences were found in emergency hospital admissions between the two periods (Table 1).

Regarding telephone care provided by the Unit, we noted a nonsignificant increase in the number of calls made by the Unit during the pandemic (P=.06). However, there was a significant increase in telephone calls made by caregivers to the Unit during the pandemic compared to the pre-pandemic period (P=.003) (Table 2).

There were no significant differences between the mean length of hospital stays (measured in days) before and during the pandemic.

Tables 1 and 2 display the variables related to the health care provided.

Significant differences were seen in the visual analogue scale (VAS) of the EuroQol five-dimension (EQ-5D) quality of life test of the primary caregivers at 12 months compared with baseline, with an increase in VAS scores at 12 months (P=.03).

The Rosenberg self-esteem scale scores of the primary caregivers were significantly lower at 3 months than at baseline (P=.03). There was a significant negative correlation between the short Zarit Caregiver Burden Interview (ZARIT) scale for primary caregiver burden at 6 months and the number of siblings (adjusted R²=0.37; P=.009). The VAS score of the EQ-5D quality of life scale for primary caregivers at 12 months correlated negatively and significantly with age at diagnosis of the child’s illness (adjusted R²=0.24; P=.04).

The difference in the index value of the EQ-5D quality of life scale for primary caregivers from baseline to 3 months was negatively correlated with the age at patient admission (adjusted R²=0.97; P=.007).

In the analysis of the relationship between caregiver quality of life and the different types of CCCs, the difference in index value of the EQ-5D quality of life scale for primary caregivers from baseline to 3 months was negatively correlated with neurological disease (adjusted R²=0.89; P=.03).

Table 3 shows the results of the tests at baseline and at 3, 6, and 12 months.

The development of telemedicine programs has transformed care for patients with chronic illnesses or those in palliative situations. Several studies including the one by Prabhakaran et al [35], highlights the effectiveness of a mobile health intervention in primary care settings within rural Indian communities. This initiative focused on preventing and managing cardiometabolic conditions, depression, and related risk factors among adults. In pediatrics, evidence-based studies, including the consensus developed by the Italian Societies of Telemedicine, Preventive and Social Pediatrics, and Pediatric Primary Care, among others, have demonstrated the critical role of telemedicine in managing patients with chronic conditions (eg, cardiology, respiratory, or neurology) or those requiring palliative care. These findings underscore the potential of telemedicine to enhance healthcare delivery by facilitating improved collaboration among multidisciplinary professionals and patients, creating innovative opportunities to optimize clinical outcomes and service quality [36].

In our study, the most common condition was neurological, which is the most frequent condition as described in literature indicating that neurological patients are the most prevalent. Our findings are in accordance with prior research, with neurological CCCs being the most frequent, followed by gastrointestinal and respiratory CCCs. Also coinciding with published findings, the mother most often fulfilled the role of the primary caregiver [13,37-41].

During the COVID-19 pandemic, as seen in other studies, a decrease was observed in both scheduled and urgent care for children with CCCs, similar to trends in the general pediatric population and among children with special health care needs [42-49].

The increase in telephone consultations during the pandemic, both initiated by the Unit and caregivers, especially the increase in calls from caregivers, may be explained by the decrease in face-to-face care in primary care and in specialized hospital care during this period. This finding highlights the support provided by the Unit via telephone to minimize the impact of the pandemic on children with CCCs, as described in several studies focused on specific care strategies aimed at this particularly vulnerable population [50].

In our study, the absence of significant differences in scheduled or urgent hospital admissions, has been described in other studies on telemedicine in children with CCCs [22]. This may be due to the high complexity of the patients included in this study, where pandemic-related factors may not have influenced the number of hospital admissions or the length of hospital stays.

Several studies have described the importance of the psychosocial needs of children with CCCs and their families and how these needs, especially concerning mental health increased during the pandemic [51,52]. To date, few studies have demonstrated improvements in the quality of life of caregivers from telemedicine programs, often due to the cross-sectional design of studies without multiple cut-off points. Our study highlights the importance of conducting studies that monitor the impact and benefits of these programs throughout the intervention to verify their usefulness [53-55].

In our study, a greater burden on primary caregivers was detected in families with more children, a demographic factor not previously described and one that should be considered to reinforce support for these families.

The quality of life of the primary caregivers measured by the VAS value of the EQ-5D scale at 12 months was poorer in those with children with CCCs diagnosed at an older age. Similarly, we found a negative correlation between the difference in the EQ-5D quality of life scale of the primary caregivers at 3 months, compared to baseline in children with CCCs who were older at admission to the Unit. Our analysis revealed that both the child's age at diagnosis and age at Unit admission were inversely associated with parental quality of life, showing the most pronounced negative impact at 3- and 12-month follow-up during the pandemic. The influence of the age of the child with a CCC on the quality of life of the primary caregivers during the pandemic has not been examined previously and merits further investigation as a potential modulator of the quality of life of the parents.

Published studies on the impact of neurological diseases have shown an increased family, work, and economic burden on primary caregivers, specifically on caregivers of children with cerebral palsy [56]. In our study, a negative correlation was found between the index value of the EQ-5D quality of life scale of the primary caregiver at 3 months, compared to baseline with neurological CCCs, which was also the most frequent CCC observed.

The observed lower self-esteem among the primary caregivers at 3 months of the telemedicine program during the pandemic setting may be explained by the effects of full confinement, including reduced psychosocial and family support, adaptation of home care with a change in care support, and the increased care burden. This reduction in the quality of life of the primary caregiver at 3 months has not been evaluated in other studies and may be useful for reinforcing psychosocial support to families, considering this period as a turning point in caregiving. Conversely, the observed improvement in VAS of the EQ-5D quality of life test may reflect an increased quality of life at 12 months, which could be attributed to adaptation to the situation as well as decreased isolation measures one year after the pandemic.

It is important to leverage the growth of the ICTs during the pandemic for the benefit of patients, especially those who are most vulnerable and with specific care needs such as children with CCCs. Telemedicine provides equity in care and efforts should be made to evaluate the impact of telemedicine programs in patients, primary caregivers, and health care teams, taking into account the clinical, economic, and quality of life variables in patients and their families monitored over time.

The use of telemedicine prior to the pandemic was demonstrated to be effective in children with CCCs, both in reducing the number of emergency department visits and costs [55,57-61]. However, in our study, it was challenging to determine with certainty whether the decrease in urgent or unscheduled care could be attributed to the COVID-19 pandemic or the telemedicine program intervention. During the pandemic, there was a decrease in emergency department visits, as many caregivers were afraid to go to hospitals to avoid infections in their children [43,45]. In addition, we must take into account the decrease in respiratory symptoms due to the confinement and isolation measures described in several studies [45,47,49]. Studies with larger and more homogeneous samples should be carried out after the pandemic to assess whether its effectiveness in terms of care is maintained without the interference caused by the pandemic.

In our study, the use of a telemedicine program during the pandemic resulted in a decrease in scheduled face-to-face care and a reduction in the number of emergency department visits compared to the prepandemic period. Conversely, we observed an increase in telephone support without any impact on hospital admissions. We identified the most vulnerable families in this group of patients, including those with a child affected by a neurological condition, those who had an older child when the disease was diagnosed, and families with a greater number of children. These insights allowed could help guide the redirection of limited resources. Our findings indicated that the telemedicine program was effective in the supporting care of children with CCCs and their families.