Challenges and Recommendations on Digital Health Sources in Pediatric Chronic Suppurative Lung Diseases

Abstract

Definition

In the context of digitalizing healthcare systems, digital health sources (DHSs) aim to enhance the efficiency, accessibility, and quality of healthcare services by leveraging technology. Multiple types of DHSs are increasingly established in healthcare, providing direct and wide communication between pediatric patients, parents, and healthcare professionals. Therefore, they are considered promising key tools to improve pediatric disease monitoring and management. At the same time, DHSs have been associated with several issues and risks, such as those related to data sharing, privacy, and the cultural readiness of the users. Yet, discussions in the literature have mostly focused on technical considerations and a user-friendly design. In contrast, the contribution of DHSs to treatment engagement in pediatric populations and data sharing has only partially been discussed. On this basis, we provide an overview of the available digital health technologies and their application in the pediatric population with chronic suppurative lung diseases; we describe the effectiveness in health-related outcomes, discuss possible challenges, and propose some recommendations that may overcome barriers in their everyday use.

1. Introduction

Chronic suppurative lung disease (CSLD) is a clinical syndrome characterized by chronic endobronchial bacterial infection and chronic productive cough. The term has been increasingly used to describe children who exhibit clinical features consistent with bronchiectasis but lack radiographic evidence of the condition [1]. Due to common features, protracted bacterial bronchitis (PBB), primary ciliary dyskinesia (PCD), cystic fibrosis (CF), and non-cystic fibrosis bronchiectasis (NCFB) are included in the CSLD spectrum clinically manifested by prolonged wet or productive cough [2]. The prevalence of pediatric CSLDs varies depending on geographic regions. In European populations, the prevalence ranges from 0.2 to 2.3/100.000, while in non-European populations, it ranges from 13.3 to 15/100.000. Among Indigenous populations in New Zealand, Australia, Alaska, and Canada, the prevalence is higher, ranging from 4.8 to 18.3/100.000. Data from Australia, Alaska, and Canada show that the peak age group prevalence for Indigenous children is around 5 years old, while data from other countries are not available [3]. Managing everyday symptoms and respiratory exacerbations is the key component of CSLD care [4]. Prompt recognition of respiratory exacerbations is crucial, as delayed recognition is linked to heightened psychological stress and a reduced health-related quality of life (HRQoL) [5]. Enhancing education for patients, parents/caregivers, and healthcare professionals (HCPs) on recognizing exacerbations and initiating additional treatments is essential [6].

In the broad context of pediatrics, digital health sources (DHSs) can provide unique opportunities for the management of a chronic disease. Children and adolescents with neurological conditions, muscular dystrophies, cerebral palsy, and behavioral disorders are some of the pediatric populations that have benefitted from the application of DHSs for monitoring symptoms, tracking physical activity, enhancing gross motor skills, and improving social interactions [7].

In pediatric chronic respiratory diseases, the DHSs in the form of digital spirometers, mHealth Apps, web-based platforms, and active video games (AVGs) have been previously applied to children with asthma and CF, mainly for monitoring symptoms, delivering health education information, or exercise programs [8,9,10,11].

Except for the possible benefits of DHS use, specific reference has been made to the technical and ethical challenges, including accuracy, privacy, security, and cyber risks [12]. DHSs should safeguard children’s rights, ensure safe use, and address pediatric-specific needs [13]. Still, technical and ethical considerations are treated as separate issues that need much further research and discussion. Thus, the literature on the pediatric CSLD population is scarce. In response to these considerations, in this review, we provide an overview of the available tools, the main functions of DHSs (remote monitoring, support, education, supervised exercise, and entertainment), and the role of these functions on health outcomes and users’ preferences. Furthermore, we discuss the main challenges of DHSs when used on the pediatric CSLD population, the health-related benefits for the users, and the benefits for the HCPs.

2. Impact of CSLD on Pediatric Population

In pediatric CSLD, lung function is significantly impaired due to recurrent inflammation, infection, and structural changes in the airways. The FEV1 declines progressively over time, while children diagnosed at older ages and with more frequent hospitalized exacerbations experience a steeper decline in FEV1 [14,15].

Reduced lung function and a high rate of hospitalizations due to exacerbations negatively impact the nutritional status of children and adolescents with CSLD. Insufficient micronutrients and increased energy requirements increase BMI abnormalities in children with NCFB [16]. BMI positively correlates with physical activity (PA) and inversely with sedentary time in children and adolescents with CF [17]. It appears that children with CF who are more active and spend less time in sedentary activities present higher BMI and better lung function than those who spend more time sedentary [17].

Along with impaired lung function and frequent exacerbations, poor management of everyday symptomatology and worse mental health are factors that negatively impact physical and exercise capacity [18,19]. Children and adolescents with CSLD experienced reduced exercise capacity due to persistent inflammation, airway damage, and recurrent respiratory infections compared to healthy peers [20].

Physical impairments also directly impact children’s and parents’ social aspects. Based on the perspectives of both children with NCFB and their parents, social burden is addressed as a result of impaired HRQoL [21,22]. Children reported difficulties keeping up with peers and participating in physical activities with others because their physical capacity was lower than their peers, and some activities triggered their symptoms (e.g., coughing) [21]. From parents’ perspective, disease severity and symptoms are those barriers that make their kids inactive and socially isolated. In an 8-week play-based therapeutic exercise program with family participation for children with NCFB, parents mentioned that this intervention can benefit children, improving their physical activity and social life through group exercise activities [23].

Children and adolescents with CSLD face not only physical impairments but also significant mental health burdens. Exacerbations and symptom severity are associated with elevated anxiety symptoms [5]. At the same time, parents of younger children with NCFB are more likely to report worse mental health than parents of children with CF [16] or healthy peers [24].

Impaired lung function, frequent exacerbations, nutritional deficiencies, and physical impairments have a total impact on school life and HRQoL in children with NCFB [5,16,25]. A multicenter pilot cohort study in children with bronchiectasis showed that the median number of days missed from school ranged from 2.5 (IQR 1.0–4.0) to 4.0 (IQR 2.0–7.0) per month [26].

Nonetheless, there are limited data on CSLD mortality in the pediatric population [3]. In England and Wales, the mortality rate in children aged <14 years is about 0.2% due to early diagnosis [27].

3. An Overview of Digital Technology in CSLD

Although this is not a systematic presentation of all possible or future applications of DHSs, we identified six (6) main sources that are currently used to serve in the health context in children and adolescents with CSLD: digital devices [28,29,30,31], mHealth apps [8,9,32,33,34], websites [35], virtual environments including active video games (AVGs) [10,36,37,38,39], serious games (SGs) [40,41], and videoconferences [11,42,43]. The aforementioned tools function mainly for remote monitoring, support, training and education, and entertainment (Table 1).

We identified remote monitoring as the basic function of DHSs, such as digital devices, mHealth apps, virtual environments including AVGs, and videoconferences. Remote monitoring is the practice of continuous data collection and serves as a fundamental function of digital health technologies used in pediatric care for respiratory diseases [44]. Smart inhalers have been developed to monitor adherence to inhaler treatments at home and emergency treatment [44]; portable spirometers replaced the traditional peak flow meters [29]; smartwatches have started to collect health data [29]; websites and mHealth apps replaced the traditional diaries used for daily symptom recording [8,9,32,34,35]; active video games and videoconference support home-based or center-based exercise programs under live supervision [11,36,37,42]. These technologies facilitate the ongoing assessment of various health parameters, such as heart rate [29], by enabling real-time data collection and providing healthcare professionals (HCPs) with vital insights into patients’ health status, treatment adherence, and overall quality of life [33,34].

Furthermore, DHSs have been found to support and empower children with CSLD and promote the development of a range of complex skills essential for improving disease knowledge [8,35], self-management [33,45], and treatment engagement [42,46]. A recent review [34] noted that DHSs empower children and their families to actively participate in managing their health issues by facilitating the recording of health behavior (e.g., physical activity) and symptoms between clinical visits (e.g., gastrointestinal problems), tracking medications, and completing previsit reports. Additionally, many DHSs offer medication reminders and appointment notifications, helping users adhere to treatment plans and manage their health more effectively [34,47]. SGs are considered computer-based games primarily designed for achieving specific health outcomes related to education or training rather than pure entertainment [48]. Serious games (SGs) and virtual environments, including active video games (AVGs) transforming respiratory therapy into a digital game, have shown promising results in increasing adherence to non-pharmacological treatment [31,41]. Studies suggest that interventions using videoconferences to support the synchronous delivery of the exercise program improve supervision by HCPs, allowing possible training modifications when necessary [11,42,43].

DHSs also offer the opportunity to embed innovations in training and education delivery. Training generally refers to the process of learning or improving specific skills (e.g., respiratory maneuvers, ACTs, exercise) through practice, instruction, or experience. Breathing and exercise training can be offered by HCPs to children through digital technologies such as AVGs, SGs, and videoconferences [10,31,36,37,42,43]. Furthermore, through AVG consoles, HCPs can modify exercise training levels according to participants’ age or exercise capacity [10,36,37].

Websites can provide specific knowledge to patients and parents/caregivers about symptom severity or recognition of exacerbations, which are important coefficients for proper disease management [35]. Educational strategies that inform HCPs and parents about the importance of recognizing and managing chronic wet cough may help earlier diagnoses and, in some cases, may even prevent the progression of CSLD disease [35]. Furthermore, mHealth apps (GENIA, MyCyFAPP, LungHealth) can deliver evidence-based education and information regarding CSLD (e.g., lung function, diagnosis, self-management, healthy lifestyle, nutrition) through interactive images, audio, and quizzes [8,34,45].

To some extent, digital games actively engage children in their health management through a playful setting. Such games may include respiratory therapy through storytelling and interaction, and they collect data regularly on the therapy [40]. However, airflow measurements taken through these games for children with CSLDs need to be validated against standard measures before the games can be implemented clinically, as has been previously conducted in other pediatric chronic respiratory diseases [49].

4. The Potential Effects of DHS’s Use in the Clinical Field

In the intervention context, the effects of DHSs focus on improving clinical parameters affecting disease progression, such as lung function, functional and exercise capacity, respiratory and peripheral muscle strength, and HRQoL.

Lung function can be improved by using digital spirometers [31] or performing exercise programs through AVGs [36] in pediatric CSLDs. Children with CF improved the percent predicted of the forced expiratory volume in 1 sec (FEV1) and vital capacity (VC) values after performing respiratory maneuvers through a digital spirometer in a 3-week intervention program [31]. Similarly, improvements in FEV1, forced vital capacity (FVC), and peak expiratory flow rate (PEF) were reported in children and adolescents with PCD who followed an 8-week aerobic exercise program through AVGs (Xbox Kinect) [36].

Exercise programs that are implemented through DHSs and include aerobic and/or resistance training can play a fundamental role in enhancing functional and exercise capacity in children and adolescents with CSLDs. In particular, pediatric populations with NCFB [37], CF [10], and PCD [36] improved functional capacity compared to the control group when an exercise program delivered through AVGs for 6 to 8 weeks was added to the usual care (ACT, pharmacological treatment, nutritional supplementation). Additionally, exercise programs delivered through videoconferences during a training period of 12 weeks have effectiveness on functional capacity [11] and peak volume of oxygen uptake (VO2peak) [42] in a pediatric population with CF.

AVGs can benefit respiratory and peripheral muscle strength. Aerobic exercise training through AVGs seems to enhance the maximal inspiratory (MIP) and expiratory pressure (MEP) of the pediatric population with NCFB [37] and PCD [36], as it forces participants on greater respiratory demands. Furthermore, exercise programs through AVGs, as well as videoconferences, benefit peripheral muscle strength in children and adolescents with NCFB, CF, and PCD, improving mainly muscle strength in the lower extremities [10,36,37,42].

Rehabilitation approaches carried out through digital technologies (e.g., AVGs) seem to positively impact physical activity and HRQoL in children and adolescents with CSLDs [10,36]. In the context of exercise training, AVGs influence parameters related to treatment burden, symptoms, and social aspects of pediatric patients with CF [10] and PCD [36], improving HRQoL scores as measured through patient-reported questionnaires.

Engagement in treatment (e.g., in ACTs) is an important issue in pediatric disease management. Measuring time spent on digital spirometers combined with feedback games can provide information about the engagement of DHS use in children with CF [31].

Exploring children’s and parents’ perceptions of DHS use provides useful insight and triggers the actions required for designing DHSs that meet their demands. From the perspective of children with NCFB, the DHSs could be useful for delivering an exercise program at home [23]. For example, mHealth apps can inform about exercise parameters or demonstrate, through audiovisual material, the proper performance of an exercise program at home. Similarly, parents mentioned that digital technology tools could be an additional aid for implementing programs at home where prioritizing is difficult. They also highlight that these tools contribute to children’s independence from parents’ help [23].

5. Challenges and Recommendations for Digital Health Tools’ Use

In this overview, we have identified the available DHS tools and their further functions of DHSs in children and adolescents with CSLDs thanks to their ability for constant and personal data collection, to entertain and engage this population in more physical activity, to support pharmacological and non-pharmacological therapy, and to share disease education issues. Despite the potential benefits, several challenges need to be considered before the widespread clinical use of DHSs.

More importantly, technical, ethical, and socioeconomic challenges are under research and discussion.

Any digital health device needs to be easy to use to avoid unnecessary stress in children and/or caregivers and to accurately monitor and transform data to HCPs [50]. On this basis, parents/patients may be convinced of the benefits of using the device and can eventually trust and use the technology. For instance, although parents and children show a preference for face-to-face exercise programs, they suggest that DHSs can be useful for completing home exercise sessions [23].

Users with more digital literacy and socioeconomic resources are disproportionately advantaged in accessing the benefits of the use of a DHS [51]. Therefore, for users with low financial resources, any DHS should be affordable, not too expensive, and difficult to access [52]. This fact should be acknowledged when HCPs propose any device for everyday use and consider the cost-effectiveness of this approach. Furthermore, healthcare systems should support fully or partially in terms of cost.

In our opinion, an important challenge is the adherence of children to treatment. When DHSs are designed based on robust research strategies and according to users’ needs, they should be implemented in the clinical setting and tested for health outcomes.

Recent strategies [53] raise awareness about the clinical needs expressed by parents/patients with bronchiectasis. Among the highest clinical needs were access to a personalized action management plan to optimize disease status and symptoms management (wet cough, sputum) as well as access to physiotherapy interventions (mostly on ACTs). More specifically, one global web-based survey of 225 parents/carers and young people with bronchiectasis from 21 countries, led by the European Lung Foundation [53], revealed that their three highest clinical needs were “having an action plan for exacerbations”, “having access to physiotherapy services and being taught on airway clearance techniques and how to use the equipment at home”, and “good communication with HCPs”. Looking at these three high-priority needs, one can realize that all can be partially managed and delivered through digital interventions, such as videoconferences, websites, and mHealth apps [54].

Furthermore, wearable devices for auscultation and remote transmission data, AVGs that aim not only to engage users in non-pharmacological interventions (e.g., ACTs) but also that transfer data to HCPs (e.g., use frequency, duration) may be some promising solutions that can lead to timely interventions and possibly to prevent the development of more serious conditions (e.g., an exacerbation) [55]. Artificial Intelligence (AI) tools are increasingly developed and used in healthcare. For example, in pediatric asthma, digital health assistants using AI technology are potentially suitable tools aiming to minimize the burden of disease monitoring [56]. Parental perception of AI integration in healthcare and decision-making regarding their children is variable. Relatively new data have documented rather good support for this approach in imaging, emergency pediatrics, and primary care, suggesting that AI is generally accepted in the medical field. A blend of AI and human supervision could prevent possible errors [57]. However, no data are available for the pediatric CSLD population. RCTs are required to identify any health outcomes most likely to benefit from these interventions.

Ethically speaking, HCPs should keep in mind that digital health technology needs to be openly and critically discussed to ensure that those who choose or cannot be part of this approach will not be unequally treated or lose access to other health services.

6. Conclusions

The use of DHSs for supporting pharmacological and non-pharmacological interventions in children and adolescents with CSLD has several promises and benefits for more multidisciplinary disease management and personalized medicine. Various digital devices, websites, mHealth applications, active video games, and serious games may facilitate remote monitoring, disease knowledge support, exercise training, and breathing exercises. Many challenges of digital technology in health use can be addressed only partially through technical solutions, whereas their ethical, social, and health-related impact needs to be identified in future research.

Author Contributions

Conceptualization, E.A.K., A.M. and V.S.; investigation of the literature, A.M., V.S. and D.M.; writing—original draft preparation, E.A.K., A.M., D.M. and V.S.; writing—review and editing, E.A.K. and K.D.; supervision, E.A.K. and K.D.; project administration, E.A.K. and K.D. All authors have read and agreed to the published version of the manuscript.

Institutional Review Board Statement

Not applicable.

Informed Consent Statement

Not applicable.

Data Availability Statement

Not applicable.

Conflicts of Interest

The authors declare no conflicts of interest.

Footnotes

Disclaimer/Publisher’s Note: The statements, opinions and data contained in all publications are solely those of the individual author(s) and contributor(s) and not of MDPI and/or the editor(s). MDPI and/or the editor(s) disclaim responsibility for any injury to people or property resulting from any ideas, methods, instructions or products referred to in the content.

Table

Table 1

Summary of the main functions served by digital health sources for health, with examples of measurements and health outcomes.

Available Digital Health Tools	Main Functions	Examples of Measurements	Health Outcomes	References
Digital devices(e.g., inhalers and inhaler add-ons, smart inhalers, digital spirometers, smartphone-connected spirometers, and smartwatches)	Remote monitoring	Physical activity (step count) Heart rate SleepLung function (FVC, FEV1)	Improve lung functionAdherence to treatment Quality of life	[28,29,30,31]
mHealth apps(e.g., Lung Health App, MyCyFAPP, Genia)	Remote monitoring Evidence-based health education/informationSupport in pharmacological and non-pharmacological treatment Communication with HCPs	Respiratory symptoms Gastrointestinal symptoms Track medicationsManagement of therapeutic regimen	Self-managementDisease knowledgeQuality of life	[8,9,32,33,34]
Websites(e.g., Child-BEAR net, Cystic Fibrosis Trust, Bronchiectasis Toolbox)	Knowledge supportwith evidence-based health education	N/A	Disease knowledge	[35]
Virtual environment (e.g., Active video game aerobic exercise, breathing video game exercises)	Remote monitoring Supervised support by HCPsEntertaining	Heart rateFatigue perception	Lung function (FEV1%, FVC%, %PEF)Respiratory muscle strength (MIP, MEP), Peripheral muscle strength (QMS, HJT, HG), Functional capacity (6MWD, ISWD, mSWT), Balance (PST, LOST, CTSIB), ADL (Glittre ADL test), HRQoL (CFQ-R, PCD-QoL)	[10,36,37,38,39]
Serious Games(e.g., Spirometer Games, Breathing Games)	Breathing training Supervised supportEntertaining	Ν/A	Compliance with ACTs	[40,41]
Videoconference(Web-based platforms) (e.g., supervised exercise)	Remote monitoring Exercise training	Heart rate	Lung function (FEV1%, FVC%),Peripheral muscle strength, Exercise capacity (CPET),Functional capacity (6MWD),Body composition (%Fat, FFM, FFMI),Anxiety and depression (RCADS)	[11,42,43]

Abbreviations: 6MWD: 6 min walking test distance; ACTs: airway clearance techniques; ADL: activities of daily living; CFQ-R: cystic fibrosis questionnaire-revised; CPET: cardiopulmonary exercise test; CTSIB: clinical test of sensory integration of balance; FEV1: forced expiratory volume in 1 sec; FFM: fat-free mass; FFMI: fat-free mass index; FVC: forced vital capacity; HCPs: healthcare professionals; HG: handgrip; HJT: horizontal jump test; LOST: limits of stability test; MEP: maximal expiratory pressure; MIP: maximum inspiratory pressure; N/A: not available; PCD: primary ciliary dyskinesia; PEF: peak expiratory flow; PST: postural stability test; QMS: quadriceps muscle strength; QoL: quality of life; RCADS: anxiety and depression scale in children-revised.

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