The Efficacy of Internet-Based Interventions in Family-Centered Empowerment Among Children with Chronic Diseases: A Mixed-Methods Systematic Review

Figures & data

Figure 1 PRISMA Flow diagram. Adapted from Page MJ, McKenzie JE, Bossuyt PM, et al. The PRISMA 2020 statement: an updated :guideline for reporting systematic reviews. BMJ. 2021;372:n71. Creative Commons.

Table 1 Quality Assessment of Included Studies Using the Mixed Methods Appraisal Tool (MMAT) Version 2018

Quantitative non-randomized studies	Are the participants representative of the target population?	Are measurements appropriate regarding both the outcome and exposure/intervention?	Are there complete outcome data?	Are the confounders accounted for in the design and analysis?	During the study period, is the intervention/exposure administered as intended?	Score (Percentage)
Wakimizu et al (2022)^Citation30	Y	Y	Y	N	Y	4/5 (80%)
Pecoraro et al (2021)^Citation31	Y	Y	Y	N	Y	4/5 (80%)
Quantitative randomized controlled trials	Is randomization appropriately performed?	Are the groups comparable at baseline?	Are there complete outcome data?	Are outcome assessors blinded to the intervention provided?	Did the participants adhere to the assigned intervention?
Swallow et al (2014)^Citation32	Y	Y	Y	N	Y	4/5 (80%)
Mixed methods studies	Is there an adequate rationale for using a mixed methods design to address the research question?	Are the different components of the study effectively integrated to answer the research question?	Are the results adequately brought together into overall interpretations?	Are divergences and inconsistencies between quantitative and qualitative results adequately addressed?	Do the different components of the study adhere to the quality criteria of each tradition of the methods involved?
Harris et al (2015)^Citation33	CT	Y	Y	N	Y	3/5 (60%)
Qualitative studies	Is the qualitative approach appropriate to answer the research question?	Are the qualitative data collection methods adequate to address the research question?	Are the findings adequately derived from the data?	Is the interpretation of results sufficiently substantiated by data?	Is there coherence between qualitative data sources, collection, analysis and interpretation?
Adriana et al (2014)^Citation34	Y	Y	Y	Y	Y	5/5 (100%)
Jones et al (2022)^Citation35	Y	Y	Y	Y	Y	5/5 (100%)

Abbreviations: Y, Yes; N, No; CT, Cannot tell.

Table 2 Characteristics of Quantitative Studies and Mixed-Method Study (n = 4)

Study	Desain	Location	Setting	Population	Age		Sample		Education		Occupation		Intervention	Model
					Children	Family (Mth/Fth)	Children	Family (Mth/Fth)	Mth	Fth	Mth	Fth
Wakimizu et al (2022)^Citation30	Non-randomize, waitlist-control trial	Japan	Home	Children with physical, intellectual, or developmental disabilities	2–20 years	20–59	55	55	Junior High School = 1, Senior High School = 7, Vocational School = 16, Bachelor’s Degree = 31.	NA	Full-time employee = 9, Self-employed = 5, Part-time employee = 20, Homemaker (Housewife) = 16, Others = 5.	NA	Online	Group-based online intervention program
Pecoraro et al (2021)^Citation31	Retrospective study	Italy	Home	Children and adolescents with obesity	5–18 years	NA	82	82	NA	NA	NA	NA	Online	Telehealth counselling
Swallow et al (2014)^Citation32	RCT	United Kingdom	Home	Children with CKD	0–19 years	NA	30	41	NA	NA	NA	NA	Online	OPIS
Harris et al (2015)^Citation33	A longitudinal, mixed-method	United Kingdom	Hospital	Children with LLI (cerebral palsy, intractable epilepsy, metabolic and genetic disorder)	Three months to 21 years	NA	32	24 mother Four father Four both parents	NA	NA	NA	NA	Online	MyQuality

Abbreviations: LLI, life-limiting illness; CHD, congenital heart disease; Mth, mother; Fth, father; EMR, electronic medical record; RCT, randomized control trial; CKD, chronic kidney disease; OPIS, the online parent information and support.

Table 3 Summary of Findings of Quantitative Studies and Mixed-Method Study (n = 4)

Domain	Number of Studies (N)	Sample Size (N)	Percentage (%)
Overall studies	4	210	100
Desain
Quantitative RCT	1	41	19.52
Quantitative non-randomized	2	137	65.24
Mixed-Method	1	32	15.24
Location
European	3	155	73.80
Asian	1	55	26,20
Setting
Home	3	178	84.76
Hospital	1	32	15.24
Intervention Model
Online	4	210	100

Abbreviation: randomized control trial, (RCT).

Table 4 Intervention Model and Outcomes of Quantitative Studies and Mixed-Method Study (n = 4)

Studi	Model	Length of Study	Follow-Up	Duration of Intervention	Instrument of Family Empowerment	Procedure of Intervention	Measurement of Outcome	Outcome
								Domain	Result
Pecoraro et al (2021)^Citation31	Telehealth counselling	Three months	Three weeks	30 minutes	NA	Parents and children participated in video calls in 3 phases, each lasting 30 minutes. Phase 1 involved discussing with a nutritionist to enhance compliance and create a nutrition plan. Phase 2 included discussing with a psychologist to provide motivational support and boost self-efficacy for children and the family. Phase 3 discussed the children’s physical activities with a kinesiologist. Additionally, video tutorials were provided on exercise types tailored to the age and physical characteristics of the children.	Anthropometry	BMI	Increase significantly (+1.2 kg/m2)
							Bioelectrical impedance anal	Fat Free Mass	Increase significantly (+3.8 Kg)
							Measuring tape	WC	Waist Circumference does not change both groups
Wakimizu et al (2022)^Citation30	Group-based online intervention program	Five month	One month	Every Sunday	The Japanese version of FES	This activity consists of 5 sessions. Session 1 (Understanding the child’s situation), Session 2 (Reflecting on the child’s and family’s situation and desires), Session 3 (Setting goals for the family and child), Session 4 (Reflecting on the activities undertaken to achieve the goals), Session 5 (small Focus Group Discussion - FGD).	FES Includes FA and SS	Total score	The FES total score was significant, with a medium effect size of 0.176, and average 7.9
							FES Includes FA and SS	FA	Scores for FA were significant, with a medium effect size of 0.128, and average 1.8
								SS	Scores for SS were significant, with a medium effect size of 0.183, and average 2.8
								SP	Scores for SS were significant, with a medium effect size of 0.175, and average 3.3
Swallow et al (2014)^Citation32	OPIS	20 weeks	On the week 20	NA	FES	The Intervention Group received support and access to the OPIS website.	REALM	Adult Literacy	Improvement in the perception of competence in managing the child’s condition.
							FaMM	Family Management	The Condition Management Ability Scale scored 44.5 for the intervention group compared to 41.9 for the control group.
							FES	Family Empowerment	The average FES score for the control group was 4.3, while it was 4.1 for the intervention group.
							DAADS	Dads Active Support	In the control group, the Amount score was 84.4, and the Helpfulness score was 69.9, whereas in the intervention group, the Amount score was 73.9, and the Helpfulness score was 76.6.
Harris et al (2015)^Citation33	MyQuality	Ten month	Three month	NA	FES	Parents were provided with direct demonstrations and training in using the application and encouraged to use it. The continuous support from MyQuality included local nurses available for the families.	FES	Seizures, constipation, pain, and sleep problems	The average FES score increased from 3.45 to 3.85, indicating a significant improvement (p≤0.01) overall. Out of 32 families, 23 chose to use the website. The most commonly reported issues were seizures (24/32), pain (6/32), sleep problems (6/32), and constipation (9/32).

Abbreviations: FES, family empowerment scale; FA, family (internal) relationships; SS, relationships with service systems; SP, involvement with the community; BMI, body mass index; IQR, inter-quartile range; WC, waist circumference; FFM, fat free mass; FM, fat mass; OPIS, the online parent information and support; DADS, the dad’s active disease support scale; FaMM, the family management measure; REALM, rapid estimate for adult literacy in medicine; FDG, focus group discussion.

Table 5 Summary of Findings of Quantitative Studies and Mixed-Method Study (n = 4)

FES Total Score	Mean Change	Mean Change Total
Pecoraro et al (2021)^Citation31	NA	4.13
Wakimizu et al (2022)^Citation30	7.9
Swallow et al (2014)^Citation32	4.1
Harris et al (2015)^Citation33	0.4

Abbreviation: FES, Family empowerment scale.

Table 6 Characteristics of Qualitative Studies and Mixed-Method Study (n = 3)

Study	Desain	Location	Setting	Population	Age		Sample		Education		Occupation		Model	Intervention
					Children	Family (Mth/Fth)	Children	Family (Mth/Fth)	Mother	Father	Mother	Father
Harris et al (2015)^Citation33	A longitudinal, mixed-method	UK	Hospital	Children with LLI (cerebral palsy, intractable epilepsy, metabolic and genetic disorder)	Three months to 21 years	NA	32	24 mth Four fth Four both parents	NA	NA	NA	NA	Online	MyQuality
Adriana et al (2014)^Citation34	Phenomenological study	US	Home	Children with ALGS	Six months to 17 years	31 to 53	16	16	College educated (14) Non college educated (2)	NA	NA	NA	Online	Facebook, YouTube, Google
Jones et al (2022)^Citation35	Observational study	Australia	Hospital	Children with CHD	4–17 years	NA	24	24	NA	NA	NA	NA	Online	EMR

Abbreviations: ALGS, Alagille syndrome; LLI, life-limiting illness; CHD, congenital heart disease; Mth, mother; Fth, father; EMR, electronic medical record.

Table 7 Interventions Model and Outcomes of Qualitative Studies and Mixed-Method Study (n = 3)

Study	Model	Length of study	Follow-up	Duration	Instrument	Process of Intervention	Outcomes
							Theme	Statement of Family
Harris et al (2015)^Citation33	MyQuality	Ten month	Three month	NA	FES	Parents were provided with direct demonstrations and training in using the application and invited to use it. MyQuality’s continued involvement included local nurses.	Acceptability	The application is easy and convenient, making care management more organized and helpful.
							Empowerment	Parents expressed experiencing an improvement in their abilities and confidence, enabling them to share information and communicate about the issues their child is facing due to the illness.
							Communication	Parents who have used the application feel that it has been helpful to them and provided access to information.
							Clinical value	The application helps improve understanding of the child’s illness and contributes to clinical management.
Adriana et al (2014)^Citation34	Facebook, YouTube, Google	NA	NA	NA	The Chronic Sorrow Questionnaire– Caregiver Version	NA	Connectedness	Mothers indicated that they use online communication for various purposes. They often initiate searches using search engines like Google and eventually access information, enhance their knowledge and empowerment, help their child, and build relationships with others with similar experiences.
							Online triggers	Online communication can trigger chronic sadness and unpleasant feelings, as they often encounter negative information about ALGS.
							Empowerment	Mothers know the value of online information sources in enhancing their knowledge, information, and empowerment in managing their child’s illness. This includes interacting with specialists, interpreting diagnoses, understanding medical instructions or recommendations, and communicating with service providers.
							Seasons of online communication	Mothers tend to use online communication more during challenging times. Additionally, mothers’ choices and frequency of online communication are based on their previous experiences and knowledge of what is suitable for their situation.
Jones et al (2022)^Citation35	EMR	Ten month	Ten month	One hour	Kuesioner family’s experience with PSM	Education includes: - How warfarin works, its indications, and factors affecting stable therapy. - Individual target therapy and dosing. - Administration of warfarin doses. - Determining when to undergo INR testing. - When to contact healthcare services. - Managing high INR at home (>5.0). - Discussion of low weight for subtherapeutic INR.	Gaining of knowledge	Parents stated that they have a basic knowledge of warfarin management, which helps them make dosing decisions at home.
							Trust and responsibility	Families are grateful for the trust and responsibility given to them, which makes them confident in their ability to manage their child’s warfarin independently.
							Saving time	The Warfarin PSM program helps them save time and reduces the time spent on decision-making.
							Availability of resources	Parents feel independent and empowered to make decisions for their children and feel supported if they need assistance or advice.

Abbreviations: ALGS, Alagille syndrome; FES, family empowerment scale; PSM, patient self-management; INR, international normalized ratio; EMR, electronic medical record.

Table 8 Summary of Findings of Qualitative Studies and Mixed-Method Study (n = 3)

Study	Intervention	Aims	Results
Harris et al (2015)^Citation33	MyQuality	To improve the Quality of Life (QoL) of children with LLI	Acceptance and ease of use of the website, as well as the graphical record of changes over time, to support ongoing management and collaborative review of medical interventions, nursing care, or social interventions.
Adriana et al (2014)^Citation34	Facebook, YouTube, Google	To explore the experiences of mothers of children with ALGS in using online health communication to manage their chronic sorrow	Online communication significantly influences how mothers of children with ALGS manage their chronic sorrow or unpleasant feelings. Findings suggest that mothers need emotional support and assistance accessing appropriate online resources.
Jones et al (2022)^Citation35	EMR	To determine the suitability and acceptance of warfarin PSM in children using the Epic Patient Portal	The average INR time was increased during PSM. Furthermore, there were no adverse events reported.

Abbreviations: ALGS, Alagille syndrome; QOL, quality of life; LLI, life limiting illness; PSM, patient self-management; INR, international normalized ratio; EMR, electronic medical record.

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