Low enrollment within pediatric research increases the cost of research, decreases generalizability, and threatens to exacerbate existing health disparities. To assess barriers and facilitators to pediatric research participation and evaluate differences by enrollment status.
Data Sources include PubMed, Embase, PsycInfo, Cumulative Index to Nursing and Allied Health Literature, and Web of Science. Study selection include peer reviewed articles that contained information related to facilitators and barriers to the parental decision whether to enroll their child in research and included the views of parents who declined. We extracted barriers and facilitators to research, enrollment status, and study characteristics, including study design, quality, and patient population.
Seventy articles were included for analysis. Facilitators of participation included: benefits, trust, support of research, informational and consent related, and relational issues. Common facilitators within those categories included health benefit to child (N = 39), altruism (N = 30), and the importance of research (N = 26). Barriers to participation included: study-related concerns, burdens of participation, lack of trust, general research concerns, informational and consent related, and relational issues. Common barriers within those categories included risks to child (N = 46), burdens of participation (N = 35), and the stress of the decision (N = 29). We had a limited ability to directly compare by enrollment status and no ability to analyze interactions between facilitators and barriers. We only included studies written in English.
This review identified key facilitators and barriers to research participation in pediatrics. The findings from this review may guide researchers aiming to create interventions to improve the parental experience of recruitment for pediatric studies and to optimize enrollment rates.
Recruitment difficulties are common within clinical research, including within pediatrics. Enrollment challenges are documented within pediatric psychiatry,1 pediatric pain medicine,2 pediatric surgery subspecialties,3 neonatology,4,5 and in rural pediatric settings.6 Recruitment within pediatrics has additional challenges. Parents often serve as proxy decision-makers for children who cannot consent7,8 and the vulnerability of sick infants and children may be perceived differently than the vulnerability of adult patients.9 Many pediatric trials have enrollment windows that are very short and occur during times of high stress for children and parents.10–12
Low enrollment rates negatively impact clinical research in a variety of ways. Low enrollment increases the cost of conducting research13 and decreases generalizability because those who enroll are different from those who do not enroll in research. Rich and colleagues found that there were differences in baseline clinical characteristics by enrollment status, with sicker babies being less likely to be enrolled in neonatal research.14 Challenges recruiting under-represented populations for research have been well documented in multiple disciplines,15 and lower enrollment rates for Black infants, compared with white infants, have been found within some neonatal clinical trials.16,17 Lower participation of under-represented groups threatens to exacerbate existing pediatric health disparities.18,19
Information about how parents decide whether to enroll their child in clinical research is lacking. Data are often hypothetical (eg, a study that asks parents if they would have enrolled their child had they been asked to participate in a clinical trial), which may not be generalizable.20 Much relevant work has struggled to include the views of those who decline,21,22 though a few manuscripts have successfully included the views of decliners.16,23–25 Learning from parents who decline participation in pediatric research is critical if we aim to have a more representative research population and subsequently a more valid and generalizable evidence base. Therefore, we identified the need for a review of published manuscripts that describe the views of parents who declined pediatric research.
Prior reviews in this area reported only subsets of pediatric trials. Tromp evaluated motivating and discouraging factors to research participation but was exclusive to drug trials.26 Fisher’s narrative synthesis reported only qualitative studies.27
In this manuscript, we focus on facilitators and barriers to pediatric research participation. We also report differences in these facilitators and barriers by enrollment status. Our review adds to the literature in at least 3 ways. First, it only includes parents who were genuinely asked to enroll their child in research. Second, it only includes studies that obtained the views of research decliners. These choices were made to increase external validity, as studies reporting the views of parents asked hypothetical questions about research and studies reporting views only of parents who chose to participate in research may be heavily biased. Third, it includes studies regardless of methodology, which enables a broader picture of the landscape within pediatric research.
Methods
Preferred Reporting Items for Systematic Review and Meta-analysis
Data and Search Strategy
We searched for peer reviewed articles, written in English, that assessed facilitators and barriers to participation among parents asked to participate in pediatric research. Because the views of decliners are of particular importance, we only included studies that reported the views of some parents who had declined research.
A research librarian performed the search using 5 databases: PubMed, Embase, PsycInfo, Cumulative Index to Nursing and Allied Health Literature, and Web of Science (Fig 1). Duplicate results were removed, and all titles and abstracts were reviewed for relevance. Selected articles were read and reviewed using our inclusion and exclusion criteria. Additional articles were identified through snowballing and citation tracking. The PRISMA flow diagram (Fig 2) shows the process of study selection.
Overview of databases searched, search strings, and search results of articles identified.
Overview of databases searched, search strings, and search results of articles identified.
PRISMA flow diagram of study identification, screening, selection, and inclusion.
PRISMA flow diagram of study identification, screening, selection, and inclusion.
Two authors (T.O., and J.N.) independently conducted the screening of abstracts and review of full texts for inclusion or exclusion. Any discrepancies were discussed and agreed upon through group consensus.
Inclusion and Exclusion Criteria
Studies met inclusion criteria if they were peer reviewed articles, contained information about the parental decision whether to enroll their child in research and included parents who declined to enroll their child in pediatric research. Studies published before our search date of July 8, 2020 were eligible for inclusion.
Studies were excluded if they were hypothetical studies (eg, asked “would you enroll your child in such a study?”), did not include decliners, did not include parental views, did not take place in a medical setting, or did not include empirical data.
Quality Appraisal
We used the Critical Appraisal Skills Program tool (Table 1) to assess qualitative studies30 and the Hawker tool (Table 2) to rate quality of each quantitative study.31 Following Cavolo, we assigned each category score and then assigned a summative “overall assessment” of low, moderate, or high.32 When a manuscript used mixed methods, quality was assessed based on the predominant methodology. Articles were not excluded based on quality assessment.
Qualitative Quality Assessment: Critical Appraisal Skills Programme
Appraisal Question . | Scoring . |
---|---|
Clear statement of aims | 1–3a |
Appropriate methodology | 1–3 |
Appropriate design | 1–3 |
Appropriate recruitment strategy | 1–3 |
Appropriate data collection | 1–3 |
Adequate consideration of the relationship between researcher and participants | 1–3 |
Ethical issues considered | 1–3 |
Data analysis sufficiently rigorous | 1–3 |
Clear statement of findings | 1–3 |
Was the research valuable | 1–3 |
Overall assessment | 10–30b |
Appraisal Question . | Scoring . |
---|---|
Clear statement of aims | 1–3a |
Appropriate methodology | 1–3 |
Appropriate design | 1–3 |
Appropriate recruitment strategy | 1–3 |
Appropriate data collection | 1–3 |
Adequate consideration of the relationship between researcher and participants | 1–3 |
Ethical issues considered | 1–3 |
Data analysis sufficiently rigorous | 1–3 |
Clear statement of findings | 1–3 |
Was the research valuable | 1–3 |
Overall assessment | 10–30b |
Yes = 3 points, unclear= 2 points, no = 1 point.
High = 25 to 30 points, moderate = 20 to 24 points, low = 15 to 19 points, very low = 10 to 14 points.
Quantitative Quality Assessment- Hawker
Appraisal Question . | Scoring . |
---|---|
Abstract and title | 1–4a |
Introduction and aims | 1–4 |
Method and data | 1–4 |
Sampling | 1–4 |
Data analysis | 1–4 |
Ethics and bias | 1–4 |
Results | 1–4 |
Transferability or generalizability | 1–4 |
Implications and usefulness | 1–4 |
Overall assessment | 9–36b |
Appraisal Question . | Scoring . |
---|---|
Abstract and title | 1–4a |
Introduction and aims | 1–4 |
Method and data | 1–4 |
Sampling | 1–4 |
Data analysis | 1–4 |
Ethics and bias | 1–4 |
Results | 1–4 |
Transferability or generalizability | 1–4 |
Implications and usefulness | 1–4 |
Overall assessment | 9–36b |
Good = 4 points, fair = 3 points, poor = 2 points, very poor = 1 point.
High = 31 to 36 points, moderate = 25 to 30 points, low = 20 to 24 points, very low = 9 to 19 points.
Data Extraction and Synthesis
Each manuscript included in our review, hereafter called enrollment study, reported the parental views related to enrollment within 1 or more pediatric research studies, hereafter called feeder study. We collected basic information about the feeder study (or studies) referenced by each of the included manuscripts: design, target disease, clinical setting, and geographic location.
Data collected from the enrollment study included study characteristics, number of those who enrolled and those who declined, nature of underlying parent study, and information collection methods. The main measures were barriers and facilitators to participation in pediatric clinical research with an emphasis on the reasons parents decline to enroll their child.
We noted varied terminology used by manuscripts, which sometimes have meaningful differences and sometimes do not. Here we will use “enrolled” and “declined.” We also recognize that families are heterogeneous and do not always consist of a “parent.” However, the term parent was used consistently in the literature, and we will use that term in this review as well to mean parent, guardian, or caregiver.
We double-coded the initial 10 manuscripts. Interrater reliability assessment identified substantial agreement for categorization of facilitators (κ coefficient 0.755) and almost perfect agreement for categorization of barriers (κ coefficient 0.876). Evaluation of the discrepancies revealed that the majority of differences were because of subcategory categorization rather than missed factors. As the agreement was high, we proceeded with data extraction performed by only a single person. We double coded 10% of the manuscripts to confirm consistency of our process, which revealed continued substantial to perfect agreement for categorization of items. We also flagged any manuscripts with data particularly challenging to be double-coded. Discrepancies or uncertain placements were adjudicated through group consensus.
For each facilitator or barrier to enrollment, we extracted whether an enrollment study identified the item and then whether it compared differences in that item by enrollment status (eg, enrolled versus declined).
Results
Study Selection
We identified 17 924 articles from our initial literature search. Duplicates and comment only results were removed. Articles that were reviewed based on title and abstract include 9889 articles and 293 articles were then reviewed in full, resulting in 68 articles meeting inclusion criteria. Two additional articles were added after snowballing and tracking, resulting in 70 articles included in this systematic review (Fig 2).22,24,33–99
Study Characteristics
Table 3 shows an overview of the characteristics of the 70 included studies. Most feeder studies were clinical trials (N = 47), the majority of which were drug studies. Both outpatient (N = 44) and inpatient (N = 33) feeder studies were represented, including representation of intensive care units (N = 17). Feeder studies included research from many countries. Enrollment studies included quantitative, qualitative, and mixed methodologies. Most common approaches were surveys (N = 32), interviews (N = 28), and secondary analyses of previously collected data, such as from recruitment logs (N = 15).
Overview of Included Studies
Study . | Summary . | Data Type of ES . | Quality Assessment of ES . | Design of FS . | Disease or system in FS . | Setting of FS . | Country of FS . |
---|---|---|---|---|---|---|---|
Barakat 201433 | Parents of children with cancer approached for multiple phase 3 clinical drug trials | Qualitative interview | 28 (High) | Clinical trial: drug study | Oncology | Outpatient | United States |
Bartlett 201834 | Parents of infants with SMA and parents of infants without SMA approached to participate in an observational biomarker study | Quantitative survey | 31 (High) | Observational | Neurology | Outpatient | United States |
Baxter 201235 | Parents of HLA positive infants approached to participate in a study tracking environmental determinants of T1DM | Quantitative secondary analysis | 33 (High) | Observational | Diabetes | Outpatient | United States, Finland, Germany, and Sweden |
Braga 201436 | Parents of infants with hydronephrosis approached for a trial to determine if continuous antibiotic prophylaxis would prevent UTI | Quantitative (not specified) | 31 (High) | Clinical trial: drug study | Renal | Outpatient | Canada |
Buck 201537 | Parents of children with intermittent exotropia were approached for a study comparing the effectiveness of surgery versus active monitoring | Qualitative interview and secondary analysis | 25 (High) | Clinical trial: nondrug study | Surgery and ophthalmology | Outpatient | United Kingdom |
Chantler 200738 | Parents of preschool children were approached to participate in 1 of 2 vaccine studies (meningitis or diphtheria) | Qualitative interview | 25 (High) | Clinical trial: vaccine study | General pediatrics | Outpatient | United Kingdom |
Chappuy 200639 | Parents of children with either cancer or HIV approached to enroll their child in 1 of several clinical trials | Quantitative survey | 30 (Moderate) | Clinical trial: nondrug study | Infectious diseases and oncology | Outpatient and ED | France |
Clausen 195440 | Mothers of second grade students approached to participate in a vaccine study | Quantitative survey and interview | 29 (Moderate) | Clinical trial: vaccine study | General pediatrics | Outpatient | United States |
D'Amanda 201941 | Parents of children with Fragile X Syndrome approached for 1 of several drug studies | Qualitative interview | 27 (High) | Clinical trial: drug study | Fragile X syndrome | Outpatient | United States |
Dahan 202022 | Parents of newborns in the NICU at risk for intubation approached to participate in an anesthesia trial | Quantitative interview | 30 (moderate) | Clinical trial: nondrug study | Anesthesia | NICU | France |
Dlugos 200542 | Parents of children with epilepsy approached to participate in a study on the genetic influences of epilepsy | Quantitative interview and secondary analysis | 30 (Moderate) | Observational | Neurology | Outpatient | United States |
Dreyzin 201443 | Parents of children with acute hepatic failure approached for 1 study evaluating the effectiveness of hepatocyte growth factor | Qualitative interview | 24 (Moderate) | Clinical trial: nondrug study | Gastrointestinal | Outpatient | United States |
Eiser 200544 | Parents of children with ALL approached for 4 quality of life studies | Qualitative interview | 15 (Low) | Clinical trial: drug study | Oncology | Outpatient and inpatient | United Kingdom |
Elemraid 201345 | Parents of children with pneumonia or empyema approached for 3 studies for analysis of blood, urine, and respiratory secretions | Qualitative secondary analysis | 23 (Moderate) | Observational | Pulmonary | Inpatient | United Kingdom |
Gattuso 200646 | Parents of children with cancer approached for ten nontherapeutic behavioral medicine studies | Quantitative secondary analysis | 31 (High) | Clinical trial and observational | Oncology | Outpatient and inpatient | United Kingdom |
Genetti 201947 | Parents of newborns approached to participate in a whole genomics sequencing study | Quantitative survey and secondary analysis | 32 (High) | Genome sequencing | Well newborn and genetics | Inpatient pediatric floor and NICU | United States |
Gill 201448 | Parents of children in cardiology, orthopedics, surgical day unit, or ED approached for 15 different nondrug trials | Quantitative survey | 33 (High) | Clinical trial and observational | General pediatrics, cardiology, anesthesia, surgery, and orthopedics | Outpatient, ED, inpatient, and OR11 | Canada |
Gonzalez 201649 | Parents of children approached to participate in several studies comparing 2 different treatments or procedures | Quantitative survey | 29 (Moderate) | Clinical trial: nondrug study | General pediatrics, gastrointestinal, and surgery | OR | United States |
Greenberg 201750 | Parents of children with 13 different medical conditions approached for a wide variety of interventional and non-interventional studies | Qualitative interview | 26 (High) | Observational and clinical trial: drug study | Not specified | Outpatient and NICU | United States |
Harth 199051 | Parents of children with asthma approached for a new drug study | Quantitative survey | 31 (High) | Clinical trial: drug study | Asthma | Outpatient and inpatient | Australia |
Harth 199252 | Parents of children with asthma approached for a study about behavior and personality | Quantitative personality tests | 34 (High) | Clinical trial: drug study | Asthma | Outpatient and inpatient | Australia |
Hayman 200153 | Parents of healthy newborns who had been offered enrollment in 1 of 2 SIDS trials | Quantitative survey | 31 (High) | Observational | Well newborn | Maternity unit | New Zealand |
Helgesson 200954 | Parents of healthy children approached for a longitudinal study on the environmental and genetic risk factors of T1DM | Quantitative survey | 32 (High) | Observational | Diabetes | Outpatient | Sweden |
Hoberman 201355 | Parents of children with vesicoureteral reflex approached to enroll in a drug trial | Quantitative survey | 33 (High) | Clinical trial: drug study | Vesicoureteral reflex | Outpatient and inpatient | United States |
Hoehn 200556 | Parents of neonates with congenital heart disease approached for 1 of 5 various clinical trials | Qualitative interview | 24 (Moderate) | Observational and clinical trial: nondrug study | Cardiovascular | OR | United States |
Hoehn 200957 | Parents of children with congenital heart disease approached for 1 of 3 observational studies | Qualitative interview | 22 (moderate) | Observational | Cardiovascular | Outpatient, inpatient, and OR | United States |
Howard Sharp 202058 | Parents of children approached to enroll their child in a genome study examining the feasibility of clinical genomic analysis | Quantitative secondary analysis | 34 (High) | Genome sequencing | Oncology | Outpatient and inpatient | United States |
Hulst 200559 | Parents of neonates and children approached for a nutritional study following intensive care admission | Quantitative secondary analysis | 33 (High) | Clinical trial: nondrug study | Nutritional | NICU, PICU | Netherlands |
Ingersgaard 201860 | Parents of children with ALL approached for1 of 2 drug trials | Qualitative interview | 27 (High) | Clinical trial: drug study | Oncology | Outpatient | Denmark |
Jay 200761 | Parents of healthy preschool children approached to participate in long-term vaccine protection trial | Quantitative survey | 30 (Moderate) | Observational | General pediatrics | Outpatient | United Kingdom |
Jenkins 200962 | Mothers of children with and without birth defects approached to participate in the National Birth Defect Study on genetic and environmental risks | Qualitative interview | 27 (High) | Observational | General pediatrics | Outpatient | United States |
Jollye 200963 | Parents of newborns approached to participate in 1 of 3 nonurgent clinical NICU trials | Qualitative interview | 24 (Moderate) | Clinical trial: nondrug study | Neonatology | NICU | United Kingdom |
Kick 201864 | Parents of children with T1DM autoantibodies, but with normal glucose tolerance test, approached to participate in the FR1DA insulin intervention study | Quantitative interview | 33 (High) | Clinical trial: drug study | Diabetes | Outpatient | Germany |
Korotchikova 201065 | Parents of healthy newborns approached to participate in an EEG study | Quantitative secondary analysis | 33 (High) | Observational | Well newborn | Maternity unit | Ireland |
Kumari 201966 | Parents of children with autism or intellectual disability approached to enroll in a genetic testing study | Quantitative survey | 33 (High) | Observational | Autism | Outpatient | India |
Labib 201867 | Parents of children with cancer approached to contribute samples to a biobank | Quantitative survey | 33 (High) | Observational | Oncology | Inpatient | Egypt |
Langley 199868 | Parents of healthy infants approached to enroll in 1 of 2 pertussis vaccine trials | Quantitative survey | 34 (High) | Clinical trial: vaccine study | General pediatrics | Outpatient | Canada |
Mason 200069 | Parents of infants approached to participate in 1 of several NICU trials | Quantitative interview | 32 (High) | Clinical trial: drug and nondrug studies | Neonatology | NICU | nine European countries |
Mazzocco 199970 | Parents of children with Fragile X syndrome approached for 1 of 2 genetic studies | Quantitative survey | 29 (Moderate) | Observational | Fragile X syndrome | Outpatient | United States |
Menon 201271 | Parents of children in the PICU approached for 45 different studies | Quantitative observation | 36 (High) | Observational and clinical trial | Not specified | PICU | Canada |
Menon 201472 | Parents of children with invasive lines approached for 1 study looking at clinical outcomes following ACTH stimulation | Quantitative secondary analysis | 34 (High) | Clinical trial: drug study | Renal | PICU | Canada |
Mihrshahi 200273 | Mothers with a history of asthma approached to enroll their newborn in an asthma prevention study | Quantitative secondary analysis | 35 (High) | Clinical trial: nondrug study | General pediatrics | Outpatient | Australia |
Morley 200574 | Parents of premature infants approached to enroll in multiple clinical trials | Quantitative survey | 32 (High) | Observational and clinical trial | Premature infants | NICU | Australia |
Mwangi 201775 | Mothers of healthy children approached for a malaria prevention drug trial | Qualitative interview | 30 (High) | Clinical trial: drug study | Well newborn and infectious disease | Outpatient | Tanzania |
Nieminen 201576 | Parents of children approached to enroll in a pneumococcal vaccine trial | Quantitative survey | 35 (High) | Clinical trial: vaccine study | General pediatrics | Outpatient | Finland |
Norris 201077 | Parents of children with AN or EDNOS with BMI < 17.5kg/m2 approached for an antipsychotic drug trial | Quantitative survey | 29 (Moderate) | Clinical trial: drug study | Psychiatric | Outpatient | Canada |
Pagano-Therrien 201778 | Parents of children with chronic health conditions approached for multiple trials | Qualitative interview | 29 (High) | Clinical trial and observational | Cystic fibrosis, T1DM, and HIV | Outpatient | United States |
Papaz 201279 | Parents of children with heart disease approached to donate samples to the heart center biobank registry | Quantitative secondary analysis | 33 (High) | Biorepository | Cardiovascular | Outpatient and inpatient | Canada |
Paquette 201980 | Parents of children in the PICU approached for 1 of 8 different studies | Quantitative survey | 32 (High) | Multiple feeder studies | Not specified | PICU | United States |
Read 200981 | Parents of children with cancer approached to participate in multiple cancer drug trials | Quantitative survey | 33 (High) | Clinical trial: drug study | Oncology | Outpatient | Canada |
Sammons 200782 | Parents of children with pneumonia approached to participate in a study comparing 2 different antibiotics | Quantitative survey | 29 (Moderate) | Clinical trial: drug study | Infectious disease | Inpatient | United Kingdom |
Scollon 201483 | Parents of children with solid tumors approached to participate in a whole genome sequencing study | Quantitative secondary analysis | 35 (High) | Genome sequencing | Oncology | Outpatient | United States |
Shah 201823 | Parents of premature infants approached to participate in a drug trial of EPO | Quantitative survey and interview | 36 (High) | Clinical trial: drug study | Neonatology | NICU | United States |
Shilling 200984 | Parents of children with various illnesses approached for a variety of drug studies | Quantitative interview | 34 (High) | Clinical trial: drug study | Asthma, neurology, and rheumatic diseases | Outpatient and NICU | United Kingdom |
Skinner 201185 | Parents of healthy newborns approached to enroll for genetic screening of Fragile X syndrome | Quantitative survey | 36 (High) | Genetic screening | Fragile X syndrome | Maternity unit | United States |
Snowdon 200686 | Parents of preterm infants approached to participate in 1 of 4 clinical drug trials | Qualitative interview | 27 (High) | Clinical trial: drug study | Neonatology | NICU | Canada |
Sureshkumar 201287 | Parents of children with symptomatic UTI were approached for a UTI preventing drug study | Quantitative survey | 34 (High) | Clinical trial: drug study | Urogynecology | Outpatient and ED | Australia |
Surun 201888 | Parents of children with cancer who entered palliative care were approached for a phase 1 or phase 2 drug trial | Quantitative secondary analysis | 33 (High) | Clinical trial: drug study | Oncology | Outpatient and inpatient | France |
Tait 199891 | Parents of children undergoing surgery were approached to participate in several anesthesia trials | Quantitative survey | 30 (Moderate) | Observational and clinical trial | Anesthesia | OR | United States |
Tait 200389 | Parents of children undergoing surgery were approached to participate in 1 of 18 anesthesia or surgical clinical trials | Quantitative survey and interview | 35 (High) | Observational and clinical trial: drug study | Anesthesia and surgery | OR | United States |
Tait 200490 | Parents of children scheduled for surgery approached for various anesthesia or surgical clinical trials | Quantitative survey | 33 (High) | Observational and clinical trial | Anesthesia and surgery | OR | United States |
Taylor 201592 | Parents of children approached for 40 various studies in the emergency department | Quantitative secondary analysis | 35 (High) | Observational and clinical trial | Not specified | ED | United States |
Thomas 201393 | Parents of children in the PICU approached to participate in 1 of multiple critical care studies | Qualitative interview | 29 (High) | Observational and clinical trial | Critical care | PICU | Canada |
Vecchi 201394 | Mothers of newborns approached for 1 study assessing neurodevelopment and mercury exposure | Quantitative survey | 36 (High) | Observational | Well newborn | Outpatient | Italy |
Volkening 201795 | Mothers of healthy newborns approached for a cohort study on mercury exposure and child neurodevelopment | Quantitative survey | 33 (High) | Newborn cohort study | Well newborn | Outpatient | Italy |
Ward 200996 | Parents of neonates approached for multiple clinical trials with more than minimal risk | Qualitative interview | 30 (High) | Clinical trial: nondrug study | Neonatology | PICU | United States |
Woodgate 201097 | Parents of children with cancer approached for 1 of many clinical trials | Qualitative interview | 28 (High) | Clinical trial | Oncology | Outpatient | Canada |
Woolfall 201398 | Parents of children with various illness approached to enroll in 1 of 4 clinical trials | Qualitative interview | 30 (High) | Clinical trial: drug study | Asthma, neurology, rhematic disease, and neonatology | Outpatient and NICU | United Kingdom |
Wynn 201099 | Parents of children with sickle cell disease approached to participate in a hydroxyurea drug trial | Quantitative survey | 28 (moderate) | Clinical trial: drug study | Hematology | Outpatient | United States |
Zupancic 199724 | Parents of newborns in the NICU approached to participate in 1 of 3 clinical drug trials | Quantitative survey | 33 (High) | Clinical trial: drug study | Neonatology | PICU | Canada |
Study . | Summary . | Data Type of ES . | Quality Assessment of ES . | Design of FS . | Disease or system in FS . | Setting of FS . | Country of FS . |
---|---|---|---|---|---|---|---|
Barakat 201433 | Parents of children with cancer approached for multiple phase 3 clinical drug trials | Qualitative interview | 28 (High) | Clinical trial: drug study | Oncology | Outpatient | United States |
Bartlett 201834 | Parents of infants with SMA and parents of infants without SMA approached to participate in an observational biomarker study | Quantitative survey | 31 (High) | Observational | Neurology | Outpatient | United States |
Baxter 201235 | Parents of HLA positive infants approached to participate in a study tracking environmental determinants of T1DM | Quantitative secondary analysis | 33 (High) | Observational | Diabetes | Outpatient | United States, Finland, Germany, and Sweden |
Braga 201436 | Parents of infants with hydronephrosis approached for a trial to determine if continuous antibiotic prophylaxis would prevent UTI | Quantitative (not specified) | 31 (High) | Clinical trial: drug study | Renal | Outpatient | Canada |
Buck 201537 | Parents of children with intermittent exotropia were approached for a study comparing the effectiveness of surgery versus active monitoring | Qualitative interview and secondary analysis | 25 (High) | Clinical trial: nondrug study | Surgery and ophthalmology | Outpatient | United Kingdom |
Chantler 200738 | Parents of preschool children were approached to participate in 1 of 2 vaccine studies (meningitis or diphtheria) | Qualitative interview | 25 (High) | Clinical trial: vaccine study | General pediatrics | Outpatient | United Kingdom |
Chappuy 200639 | Parents of children with either cancer or HIV approached to enroll their child in 1 of several clinical trials | Quantitative survey | 30 (Moderate) | Clinical trial: nondrug study | Infectious diseases and oncology | Outpatient and ED | France |
Clausen 195440 | Mothers of second grade students approached to participate in a vaccine study | Quantitative survey and interview | 29 (Moderate) | Clinical trial: vaccine study | General pediatrics | Outpatient | United States |
D'Amanda 201941 | Parents of children with Fragile X Syndrome approached for 1 of several drug studies | Qualitative interview | 27 (High) | Clinical trial: drug study | Fragile X syndrome | Outpatient | United States |
Dahan 202022 | Parents of newborns in the NICU at risk for intubation approached to participate in an anesthesia trial | Quantitative interview | 30 (moderate) | Clinical trial: nondrug study | Anesthesia | NICU | France |
Dlugos 200542 | Parents of children with epilepsy approached to participate in a study on the genetic influences of epilepsy | Quantitative interview and secondary analysis | 30 (Moderate) | Observational | Neurology | Outpatient | United States |
Dreyzin 201443 | Parents of children with acute hepatic failure approached for 1 study evaluating the effectiveness of hepatocyte growth factor | Qualitative interview | 24 (Moderate) | Clinical trial: nondrug study | Gastrointestinal | Outpatient | United States |
Eiser 200544 | Parents of children with ALL approached for 4 quality of life studies | Qualitative interview | 15 (Low) | Clinical trial: drug study | Oncology | Outpatient and inpatient | United Kingdom |
Elemraid 201345 | Parents of children with pneumonia or empyema approached for 3 studies for analysis of blood, urine, and respiratory secretions | Qualitative secondary analysis | 23 (Moderate) | Observational | Pulmonary | Inpatient | United Kingdom |
Gattuso 200646 | Parents of children with cancer approached for ten nontherapeutic behavioral medicine studies | Quantitative secondary analysis | 31 (High) | Clinical trial and observational | Oncology | Outpatient and inpatient | United Kingdom |
Genetti 201947 | Parents of newborns approached to participate in a whole genomics sequencing study | Quantitative survey and secondary analysis | 32 (High) | Genome sequencing | Well newborn and genetics | Inpatient pediatric floor and NICU | United States |
Gill 201448 | Parents of children in cardiology, orthopedics, surgical day unit, or ED approached for 15 different nondrug trials | Quantitative survey | 33 (High) | Clinical trial and observational | General pediatrics, cardiology, anesthesia, surgery, and orthopedics | Outpatient, ED, inpatient, and OR11 | Canada |
Gonzalez 201649 | Parents of children approached to participate in several studies comparing 2 different treatments or procedures | Quantitative survey | 29 (Moderate) | Clinical trial: nondrug study | General pediatrics, gastrointestinal, and surgery | OR | United States |
Greenberg 201750 | Parents of children with 13 different medical conditions approached for a wide variety of interventional and non-interventional studies | Qualitative interview | 26 (High) | Observational and clinical trial: drug study | Not specified | Outpatient and NICU | United States |
Harth 199051 | Parents of children with asthma approached for a new drug study | Quantitative survey | 31 (High) | Clinical trial: drug study | Asthma | Outpatient and inpatient | Australia |
Harth 199252 | Parents of children with asthma approached for a study about behavior and personality | Quantitative personality tests | 34 (High) | Clinical trial: drug study | Asthma | Outpatient and inpatient | Australia |
Hayman 200153 | Parents of healthy newborns who had been offered enrollment in 1 of 2 SIDS trials | Quantitative survey | 31 (High) | Observational | Well newborn | Maternity unit | New Zealand |
Helgesson 200954 | Parents of healthy children approached for a longitudinal study on the environmental and genetic risk factors of T1DM | Quantitative survey | 32 (High) | Observational | Diabetes | Outpatient | Sweden |
Hoberman 201355 | Parents of children with vesicoureteral reflex approached to enroll in a drug trial | Quantitative survey | 33 (High) | Clinical trial: drug study | Vesicoureteral reflex | Outpatient and inpatient | United States |
Hoehn 200556 | Parents of neonates with congenital heart disease approached for 1 of 5 various clinical trials | Qualitative interview | 24 (Moderate) | Observational and clinical trial: nondrug study | Cardiovascular | OR | United States |
Hoehn 200957 | Parents of children with congenital heart disease approached for 1 of 3 observational studies | Qualitative interview | 22 (moderate) | Observational | Cardiovascular | Outpatient, inpatient, and OR | United States |
Howard Sharp 202058 | Parents of children approached to enroll their child in a genome study examining the feasibility of clinical genomic analysis | Quantitative secondary analysis | 34 (High) | Genome sequencing | Oncology | Outpatient and inpatient | United States |
Hulst 200559 | Parents of neonates and children approached for a nutritional study following intensive care admission | Quantitative secondary analysis | 33 (High) | Clinical trial: nondrug study | Nutritional | NICU, PICU | Netherlands |
Ingersgaard 201860 | Parents of children with ALL approached for1 of 2 drug trials | Qualitative interview | 27 (High) | Clinical trial: drug study | Oncology | Outpatient | Denmark |
Jay 200761 | Parents of healthy preschool children approached to participate in long-term vaccine protection trial | Quantitative survey | 30 (Moderate) | Observational | General pediatrics | Outpatient | United Kingdom |
Jenkins 200962 | Mothers of children with and without birth defects approached to participate in the National Birth Defect Study on genetic and environmental risks | Qualitative interview | 27 (High) | Observational | General pediatrics | Outpatient | United States |
Jollye 200963 | Parents of newborns approached to participate in 1 of 3 nonurgent clinical NICU trials | Qualitative interview | 24 (Moderate) | Clinical trial: nondrug study | Neonatology | NICU | United Kingdom |
Kick 201864 | Parents of children with T1DM autoantibodies, but with normal glucose tolerance test, approached to participate in the FR1DA insulin intervention study | Quantitative interview | 33 (High) | Clinical trial: drug study | Diabetes | Outpatient | Germany |
Korotchikova 201065 | Parents of healthy newborns approached to participate in an EEG study | Quantitative secondary analysis | 33 (High) | Observational | Well newborn | Maternity unit | Ireland |
Kumari 201966 | Parents of children with autism or intellectual disability approached to enroll in a genetic testing study | Quantitative survey | 33 (High) | Observational | Autism | Outpatient | India |
Labib 201867 | Parents of children with cancer approached to contribute samples to a biobank | Quantitative survey | 33 (High) | Observational | Oncology | Inpatient | Egypt |
Langley 199868 | Parents of healthy infants approached to enroll in 1 of 2 pertussis vaccine trials | Quantitative survey | 34 (High) | Clinical trial: vaccine study | General pediatrics | Outpatient | Canada |
Mason 200069 | Parents of infants approached to participate in 1 of several NICU trials | Quantitative interview | 32 (High) | Clinical trial: drug and nondrug studies | Neonatology | NICU | nine European countries |
Mazzocco 199970 | Parents of children with Fragile X syndrome approached for 1 of 2 genetic studies | Quantitative survey | 29 (Moderate) | Observational | Fragile X syndrome | Outpatient | United States |
Menon 201271 | Parents of children in the PICU approached for 45 different studies | Quantitative observation | 36 (High) | Observational and clinical trial | Not specified | PICU | Canada |
Menon 201472 | Parents of children with invasive lines approached for 1 study looking at clinical outcomes following ACTH stimulation | Quantitative secondary analysis | 34 (High) | Clinical trial: drug study | Renal | PICU | Canada |
Mihrshahi 200273 | Mothers with a history of asthma approached to enroll their newborn in an asthma prevention study | Quantitative secondary analysis | 35 (High) | Clinical trial: nondrug study | General pediatrics | Outpatient | Australia |
Morley 200574 | Parents of premature infants approached to enroll in multiple clinical trials | Quantitative survey | 32 (High) | Observational and clinical trial | Premature infants | NICU | Australia |
Mwangi 201775 | Mothers of healthy children approached for a malaria prevention drug trial | Qualitative interview | 30 (High) | Clinical trial: drug study | Well newborn and infectious disease | Outpatient | Tanzania |
Nieminen 201576 | Parents of children approached to enroll in a pneumococcal vaccine trial | Quantitative survey | 35 (High) | Clinical trial: vaccine study | General pediatrics | Outpatient | Finland |
Norris 201077 | Parents of children with AN or EDNOS with BMI < 17.5kg/m2 approached for an antipsychotic drug trial | Quantitative survey | 29 (Moderate) | Clinical trial: drug study | Psychiatric | Outpatient | Canada |
Pagano-Therrien 201778 | Parents of children with chronic health conditions approached for multiple trials | Qualitative interview | 29 (High) | Clinical trial and observational | Cystic fibrosis, T1DM, and HIV | Outpatient | United States |
Papaz 201279 | Parents of children with heart disease approached to donate samples to the heart center biobank registry | Quantitative secondary analysis | 33 (High) | Biorepository | Cardiovascular | Outpatient and inpatient | Canada |
Paquette 201980 | Parents of children in the PICU approached for 1 of 8 different studies | Quantitative survey | 32 (High) | Multiple feeder studies | Not specified | PICU | United States |
Read 200981 | Parents of children with cancer approached to participate in multiple cancer drug trials | Quantitative survey | 33 (High) | Clinical trial: drug study | Oncology | Outpatient | Canada |
Sammons 200782 | Parents of children with pneumonia approached to participate in a study comparing 2 different antibiotics | Quantitative survey | 29 (Moderate) | Clinical trial: drug study | Infectious disease | Inpatient | United Kingdom |
Scollon 201483 | Parents of children with solid tumors approached to participate in a whole genome sequencing study | Quantitative secondary analysis | 35 (High) | Genome sequencing | Oncology | Outpatient | United States |
Shah 201823 | Parents of premature infants approached to participate in a drug trial of EPO | Quantitative survey and interview | 36 (High) | Clinical trial: drug study | Neonatology | NICU | United States |
Shilling 200984 | Parents of children with various illnesses approached for a variety of drug studies | Quantitative interview | 34 (High) | Clinical trial: drug study | Asthma, neurology, and rheumatic diseases | Outpatient and NICU | United Kingdom |
Skinner 201185 | Parents of healthy newborns approached to enroll for genetic screening of Fragile X syndrome | Quantitative survey | 36 (High) | Genetic screening | Fragile X syndrome | Maternity unit | United States |
Snowdon 200686 | Parents of preterm infants approached to participate in 1 of 4 clinical drug trials | Qualitative interview | 27 (High) | Clinical trial: drug study | Neonatology | NICU | Canada |
Sureshkumar 201287 | Parents of children with symptomatic UTI were approached for a UTI preventing drug study | Quantitative survey | 34 (High) | Clinical trial: drug study | Urogynecology | Outpatient and ED | Australia |
Surun 201888 | Parents of children with cancer who entered palliative care were approached for a phase 1 or phase 2 drug trial | Quantitative secondary analysis | 33 (High) | Clinical trial: drug study | Oncology | Outpatient and inpatient | France |
Tait 199891 | Parents of children undergoing surgery were approached to participate in several anesthesia trials | Quantitative survey | 30 (Moderate) | Observational and clinical trial | Anesthesia | OR | United States |
Tait 200389 | Parents of children undergoing surgery were approached to participate in 1 of 18 anesthesia or surgical clinical trials | Quantitative survey and interview | 35 (High) | Observational and clinical trial: drug study | Anesthesia and surgery | OR | United States |
Tait 200490 | Parents of children scheduled for surgery approached for various anesthesia or surgical clinical trials | Quantitative survey | 33 (High) | Observational and clinical trial | Anesthesia and surgery | OR | United States |
Taylor 201592 | Parents of children approached for 40 various studies in the emergency department | Quantitative secondary analysis | 35 (High) | Observational and clinical trial | Not specified | ED | United States |
Thomas 201393 | Parents of children in the PICU approached to participate in 1 of multiple critical care studies | Qualitative interview | 29 (High) | Observational and clinical trial | Critical care | PICU | Canada |
Vecchi 201394 | Mothers of newborns approached for 1 study assessing neurodevelopment and mercury exposure | Quantitative survey | 36 (High) | Observational | Well newborn | Outpatient | Italy |
Volkening 201795 | Mothers of healthy newborns approached for a cohort study on mercury exposure and child neurodevelopment | Quantitative survey | 33 (High) | Newborn cohort study | Well newborn | Outpatient | Italy |
Ward 200996 | Parents of neonates approached for multiple clinical trials with more than minimal risk | Qualitative interview | 30 (High) | Clinical trial: nondrug study | Neonatology | PICU | United States |
Woodgate 201097 | Parents of children with cancer approached for 1 of many clinical trials | Qualitative interview | 28 (High) | Clinical trial | Oncology | Outpatient | Canada |
Woolfall 201398 | Parents of children with various illness approached to enroll in 1 of 4 clinical trials | Qualitative interview | 30 (High) | Clinical trial: drug study | Asthma, neurology, rhematic disease, and neonatology | Outpatient and NICU | United Kingdom |
Wynn 201099 | Parents of children with sickle cell disease approached to participate in a hydroxyurea drug trial | Quantitative survey | 28 (moderate) | Clinical trial: drug study | Hematology | Outpatient | United States |
Zupancic 199724 | Parents of newborns in the NICU approached to participate in 1 of 3 clinical drug trials | Quantitative survey | 33 (High) | Clinical trial: drug study | Neonatology | PICU | Canada |
ACTH, adrenocorticotropin hormone; ALL, acute lymphoblastic leukemia; AN, anorexia nervosa; ED, emergency department; EDNOS, eating disorder not otherwise specified; EPO, erythropoietin ES, enrollment study; FS, feeder study; OR, operating room; SIDS, sudden infant death syndrome; SMA, spinal muscular atrophy; T1DM, Type-1 diabetes mellitus; UTI, urinary tract infection.
Study Quality and Risk of Bias
Overall study quality of qualitative studies, using the Critical Appraisal Skills Programme tool,30 ranged from 15 (low) to 30 (high) with most studies scoring in the high category (25–30). Overall study quality of quantitative studies, using the Hawker tool,31 ranged from 28 (moderate) to 36 (high) with most studies scoring in the high category (31–36). Quality assessment was used to better understand the overall quality of the included studies; it was not used to exclude low scoring articles. Quality assessment scores are included in Table 3.
Categorization of Barriers and Facilitators to Pediatric Research Participation
Because of heterogeneity in methodologies and terminologies, our team needed to create a schema of categories to generate meaningful comparisons between studies. This was done through literature review, prior work, and multiple rounds of reading manuscripts to reach consensus among the 3 authors. First, we distinguished between facilitators and barriers. Although some items were easy to categorize (eg, “perceived health benefit to child” as a facilitator), others were not straightforward. For example, it seemed clear that “additional travel to medical center” should be a barrier, which we included in “burdens of participation.” However, some manuscripts reported the “lack of additional burden for family” as a reason that parents cited as supporting research participation. We chose to always keep the framing of each item as presented in the manuscript, rather than reporting the inverse. See Tables 4 and 5 for our categories, the items within each category, definitions, and exemplary quotes.
Definitions of Facilitators to Pediatric Research Participation
Category . | Facilitators . | Definition . | Example . |
---|---|---|---|
Benefits | Health benefit to child | Direct health benefit to the child | To have “the most advanced treatment available” (Chappuy 2006)39 |
Non-health benefit to child | A benefit to the child outside of health | The child would be more “at ease with the home visits” provided by enrolling (Chantler 2007)38 | |
More contact with medical team | More contact or time with medical team | “Study staff contactable at any time to answer query” (Jay 2007)61 | |
Direct benefit to parent | A direct benefit to the parent | For the parent “to meet people” (Harth 1990)51 | |
Minimal burden to child or parent | Minimal burden (eg, time or pain) to child or parent | “No additional tests or hospital visits” for the child (Eiser 2005)44 | |
Financial reimbursement | Financial reimbursement | Reimbursement of travel costs (Harth 1990)51 | |
Trust | Trust in clinician | Trust in the clinician(s) | Trust in the medical team (Chappuy 2006)39 |
Trust in medical researchers | Trust in medical researchers | Trust that the researchers would not “purposely do something they think can harm babies” (Snowdon 2006)86 | |
Trust in particular institution | Trust in a particular institution (eg, hospital or university) | “Study proposed by an institution that I trust” (Vecchi 2013)94 | |
Trust in medicine in general | Trust in medicine in general | “Trust in the medical system” (Tait 2004)90 | |
Trust in research | Trust in research or the safety of research | “Trust in research” (Hoberman 2013)55 | |
Trust (other) | Trust that did not fit into another category | “Trust in vaccines” (Chantler 2007)38 | |
Support of research | Altruism | To benefit or help others | “Other children might benefit” (Tait 1998)91 |
Importance of medical research | The importance of research or the importance of contribution to research | To contribute to “medical research” (Harth 1990)51 | |
Informational and consent process related | Felt as only option | Felt as only option remaining to help the child | Parents felt that there “were no available therapeutic alternatives” (Chappuy 2006)39 |
Understanding of research materials | Understanding of research materials (eg, consent documents) | Understanding of the consent (Tait 2003)89 | |
Experience or lack of experience with research | Past experience of lack of experience with medical research, including being enrolled in a different trial at the time | “Prior experience with research” (Chappuy 2006)39 | |
Relational | Positive relationship to researcher | Positive relationship or experience with the team conducting the research | The researchers were viewed as “friendly and relaxed” (Tait 2003)89 |
Influence of family and friends | Influence of family and friends | “Felt pressure from my family or friends” (Read 2009)81 | |
Participation encouraged by clinical team | Participation was encouraged by the child's clinical team | “Doctor asked them to participate” (Sammons 2007)82 | |
Child's desire to participate | Child's desire to participate in research | “Child wanted to participate” (Paquette 2019)80 |
Category . | Facilitators . | Definition . | Example . |
---|---|---|---|
Benefits | Health benefit to child | Direct health benefit to the child | To have “the most advanced treatment available” (Chappuy 2006)39 |
Non-health benefit to child | A benefit to the child outside of health | The child would be more “at ease with the home visits” provided by enrolling (Chantler 2007)38 | |
More contact with medical team | More contact or time with medical team | “Study staff contactable at any time to answer query” (Jay 2007)61 | |
Direct benefit to parent | A direct benefit to the parent | For the parent “to meet people” (Harth 1990)51 | |
Minimal burden to child or parent | Minimal burden (eg, time or pain) to child or parent | “No additional tests or hospital visits” for the child (Eiser 2005)44 | |
Financial reimbursement | Financial reimbursement | Reimbursement of travel costs (Harth 1990)51 | |
Trust | Trust in clinician | Trust in the clinician(s) | Trust in the medical team (Chappuy 2006)39 |
Trust in medical researchers | Trust in medical researchers | Trust that the researchers would not “purposely do something they think can harm babies” (Snowdon 2006)86 | |
Trust in particular institution | Trust in a particular institution (eg, hospital or university) | “Study proposed by an institution that I trust” (Vecchi 2013)94 | |
Trust in medicine in general | Trust in medicine in general | “Trust in the medical system” (Tait 2004)90 | |
Trust in research | Trust in research or the safety of research | “Trust in research” (Hoberman 2013)55 | |
Trust (other) | Trust that did not fit into another category | “Trust in vaccines” (Chantler 2007)38 | |
Support of research | Altruism | To benefit or help others | “Other children might benefit” (Tait 1998)91 |
Importance of medical research | The importance of research or the importance of contribution to research | To contribute to “medical research” (Harth 1990)51 | |
Informational and consent process related | Felt as only option | Felt as only option remaining to help the child | Parents felt that there “were no available therapeutic alternatives” (Chappuy 2006)39 |
Understanding of research materials | Understanding of research materials (eg, consent documents) | Understanding of the consent (Tait 2003)89 | |
Experience or lack of experience with research | Past experience of lack of experience with medical research, including being enrolled in a different trial at the time | “Prior experience with research” (Chappuy 2006)39 | |
Relational | Positive relationship to researcher | Positive relationship or experience with the team conducting the research | The researchers were viewed as “friendly and relaxed” (Tait 2003)89 |
Influence of family and friends | Influence of family and friends | “Felt pressure from my family or friends” (Read 2009)81 | |
Participation encouraged by clinical team | Participation was encouraged by the child's clinical team | “Doctor asked them to participate” (Sammons 2007)82 | |
Child's desire to participate | Child's desire to participate in research | “Child wanted to participate” (Paquette 2019)80 |
Definitions of Barriers to Pediatric Research Participation
Category . | Barriers . | Definition . | Example . |
---|---|---|---|
Concerns about study participation | Risk to child | Risk to child, including side-effects and safety concerns | “Concerns about side-effects” (Buck 2015)37 |
Negative impact on medical care | Negative impact on medical care, including preference for the standard of care | “Study would interfere with the standard care” (Hoberman 2013)55 | |
Lack of direct benefit to child | Lack of a direct benefit to child | “Drug was less likely to benefit their child” (D'Amanda 2019)41 | |
Child's own medical concerns | Child's own medical concerns or knowledge | “Child does not know true diagnosis” (Chappuy 2006)39 | |
Phlebotomy concerns | Concerns with blood draws | “Blood-draw requirements” mentioned as a barrier (D'Amanda 2019)41 | |
Burdens | Logistics or burden of participation | Logistics or burdens of participation (eg, travel, time, or money) | “Excessive travel” (Bartlett 2018)34 |
Lack of trust | Lack of trust in clinicians | Lack of trust in the clinicians | “Poor confidence in physician” (Chappuy 2006)39 |
Lack of trust in medical researchers | Lack of trust in medical researchers | “Distrust of researchers” (Mason 2000)69 | |
Lack of trust in particular institution | Lack of trust in a particular institution (eg, hospital or university) | “Distrust in the hospital” (Harth 1990)51 | |
Lack of trust in medicine in general | Lack of trust in medicine in general | “Distrust in modern medicine” (Harth 1990)51 | |
Lack of trust in research | Lack of trust in research or the safety of research | “Being uncomfortable with research” (Mazzocco 1999)70 | |
General research concerns | Lack of interest in research | Lack of interest in research | “Not interested in research” (Genetti 2019)47 |
Guinea pig concerns | Concerns with being the first to experience a new intervention | Not wanting “anything tested out” on their child (Chantler 2007)38 | |
Privacy concerns | Concerns regarding privacy of the parent or child | “Loss of confidentiality” (Menon 2012)71 | |
Lack of belief in research | General lack of belief in the benefit of research; statements against the general idea of research | “Being against research” (Hulst 2005)59 | |
Discomfort with randomization | Discomfort related to randomization and blinding | “Parental unwillingness to be blinded to study” (Braga 2014)36 | |
Genetic testing concerns | Concerns regarding genetic testing | “Uncomfortable with genetic screening” (Genetti 2019)47 | |
Informational and consent process related | Not enough information provided about study; inadequate understanding of study | Not enough information provided about study or inadequate understanding of study by the parent | “Lack of information” and “unclear information” (Chappuy 2006)39 |
Not enough time to make decision about enrollment | Not enough time to make decision about enrollment | “Lack of time to make decision” (Eiser 2005)44 | |
Not enough knowledge about medical condition or lack of comfort with clinical decision | Not enough knowledge about medical condition or lack of comfort with clinical decision | “Parents did not believe their child had Fragile-X” (Mazzocco 1990)70 | |
Decision too stressful | Decision was too stressful | “Overwhelmed with research studies” (Papaz 2012)79 | |
Parental perception of illness | Parental perception of the illness of the child | “Patient too ill to participate” (Bartlett 2018)34 | |
Language Barrier | Language barrier | “Language barriers” cited as a reason for nonparticipation (Helgesson 2009)54 | |
Prior experience or lack of experience with research | Past experience of lack of experience with medical research, including being enrolled in a different trial at the time | “Past experiences with research had both positive and negative effects” (Thomas 2013)93 | |
Relational | Discomfort with proxy nature of consent | Discomfort with the parent deciding about participation in a clinical trial for their child | Being “uncomfortable making decision on behalf of the child” (Chantler 2007)38 |
One parent wanted to enroll, other did not | One parent wanted to enroll, the other did not | “Discordance in their decision making between parents” (Genetti 2019)47 | |
Family problems | Family problems (eg, divorce) | “Family or marital problems” (Mihrshahi 2002)73 | |
Feeling pressured | Feeling pressured to enroll in the trial | “Feeling pressured into taking part in the study” (Hayman 2001)53 | |
Child’s wishes related to enrollment | Child’s wishes related to enrollment | “Child’s unwillingness to participate” (Greenberg 2017)50 | |
Decision-making style | Decision-making concerns not-otherwise-specified | Decision making style: family or self versus shared with doctor (Hoberman 2013)55 |
Category . | Barriers . | Definition . | Example . |
---|---|---|---|
Concerns about study participation | Risk to child | Risk to child, including side-effects and safety concerns | “Concerns about side-effects” (Buck 2015)37 |
Negative impact on medical care | Negative impact on medical care, including preference for the standard of care | “Study would interfere with the standard care” (Hoberman 2013)55 | |
Lack of direct benefit to child | Lack of a direct benefit to child | “Drug was less likely to benefit their child” (D'Amanda 2019)41 | |
Child's own medical concerns | Child's own medical concerns or knowledge | “Child does not know true diagnosis” (Chappuy 2006)39 | |
Phlebotomy concerns | Concerns with blood draws | “Blood-draw requirements” mentioned as a barrier (D'Amanda 2019)41 | |
Burdens | Logistics or burden of participation | Logistics or burdens of participation (eg, travel, time, or money) | “Excessive travel” (Bartlett 2018)34 |
Lack of trust | Lack of trust in clinicians | Lack of trust in the clinicians | “Poor confidence in physician” (Chappuy 2006)39 |
Lack of trust in medical researchers | Lack of trust in medical researchers | “Distrust of researchers” (Mason 2000)69 | |
Lack of trust in particular institution | Lack of trust in a particular institution (eg, hospital or university) | “Distrust in the hospital” (Harth 1990)51 | |
Lack of trust in medicine in general | Lack of trust in medicine in general | “Distrust in modern medicine” (Harth 1990)51 | |
Lack of trust in research | Lack of trust in research or the safety of research | “Being uncomfortable with research” (Mazzocco 1999)70 | |
General research concerns | Lack of interest in research | Lack of interest in research | “Not interested in research” (Genetti 2019)47 |
Guinea pig concerns | Concerns with being the first to experience a new intervention | Not wanting “anything tested out” on their child (Chantler 2007)38 | |
Privacy concerns | Concerns regarding privacy of the parent or child | “Loss of confidentiality” (Menon 2012)71 | |
Lack of belief in research | General lack of belief in the benefit of research; statements against the general idea of research | “Being against research” (Hulst 2005)59 | |
Discomfort with randomization | Discomfort related to randomization and blinding | “Parental unwillingness to be blinded to study” (Braga 2014)36 | |
Genetic testing concerns | Concerns regarding genetic testing | “Uncomfortable with genetic screening” (Genetti 2019)47 | |
Informational and consent process related | Not enough information provided about study; inadequate understanding of study | Not enough information provided about study or inadequate understanding of study by the parent | “Lack of information” and “unclear information” (Chappuy 2006)39 |
Not enough time to make decision about enrollment | Not enough time to make decision about enrollment | “Lack of time to make decision” (Eiser 2005)44 | |
Not enough knowledge about medical condition or lack of comfort with clinical decision | Not enough knowledge about medical condition or lack of comfort with clinical decision | “Parents did not believe their child had Fragile-X” (Mazzocco 1990)70 | |
Decision too stressful | Decision was too stressful | “Overwhelmed with research studies” (Papaz 2012)79 | |
Parental perception of illness | Parental perception of the illness of the child | “Patient too ill to participate” (Bartlett 2018)34 | |
Language Barrier | Language barrier | “Language barriers” cited as a reason for nonparticipation (Helgesson 2009)54 | |
Prior experience or lack of experience with research | Past experience of lack of experience with medical research, including being enrolled in a different trial at the time | “Past experiences with research had both positive and negative effects” (Thomas 2013)93 | |
Relational | Discomfort with proxy nature of consent | Discomfort with the parent deciding about participation in a clinical trial for their child | Being “uncomfortable making decision on behalf of the child” (Chantler 2007)38 |
One parent wanted to enroll, other did not | One parent wanted to enroll, the other did not | “Discordance in their decision making between parents” (Genetti 2019)47 | |
Family problems | Family problems (eg, divorce) | “Family or marital problems” (Mihrshahi 2002)73 | |
Feeling pressured | Feeling pressured to enroll in the trial | “Feeling pressured into taking part in the study” (Hayman 2001)53 | |
Child’s wishes related to enrollment | Child’s wishes related to enrollment | “Child’s unwillingness to participate” (Greenberg 2017)50 | |
Decision-making style | Decision-making concerns not-otherwise-specified | Decision making style: family or self versus shared with doctor (Hoberman 2013)55 |
Qualitative Meta-summary of Facilitators
Tables 6 and 7 show the facilitators to pediatric research. In Table 6, we share the number and name of each study that included the facilitator. In Table 7, we present only studies that directly compared the facilitator between enrolled and declined parents. For quantitative comparisons, we present the relevant statistic of the differences by enrollment status.
Facilitators for Participation in Pediatric Research
Category . | Facilitators . | Number of Studies . | Individual Studies . |
---|---|---|---|
Benefits | Health benefit to child | 39 | Barakat 2014,33 Buck 2015,37 Chantler 2007,38 Chappuy 2006,39 Clausen 1954a,40 D'Amanda 2019,a,41 Dahan 2020,22 Dreyzin 2014a,43 Eiser 2005,44 Greenberg 2017,50 Harth 1990,51 Harth 1992,52 Hayman 2001,53 Helgesson 2009,54 Hoberman 2013a,55 Hoehn 2005,56 Ingersgaard 2018,60 Jollye 2009,63 Kumari 2019,66 Langley 1998,68 Mason 2000,69 Morley 2005,74 Mwangi 2017a,75 Nieminen 2015,76 Paquette 2019,80 Read 2009,81 Sammons 2007,82 Shah 2018a,23 Shilling 2009,84 Snowdon 2006,86 Tait 1998,91 Tait 2003a,89 Tait 2004a,90 Thomas 2013,93 Ward 2009,96 Woodgate 2010,97 Woolfall 2013,98 Wynn 2010,99 Zupancic 1997a,24 |
Nonhealth benefit to child | 8 | Chantler 2007,38 D'Amanda 2019,41 Helgesson 2009,54 Shilling 2009,84 Skinner 2011,85 Tait 1998,91 Thomas 2013,93 Wynn 2010.99 | |
More contact with medical team | 5 | Chantler 2007,38 Dreyzin 2014,43 Harth 1990,52 Mwangi 2017a.75 | |
Direct benefit to parent | 6 | Chantler 2007,38 Harth 1990,51 Helgesson 2009,54 Langley 1998,68 Skinner 2011,85 Ward 2009.96 | |
Minimal burden to child or parent | 16 | Buck 2015,37 Dreyzin 2014,43 Eiser 2005,44 Greenberg 2017,50 Helgesson 2009,54 Hoberman 2013a,55 Mason 2000,69 Pagano-Therrien 2017,78 Paquette 2019,80 Read 2009,81 Sammons 2007,82 Shilling 2009,84 Skinner 2011,85 Taylor 2015a,92 Tait 1998,91 Woodgate 2010.97 | |
Financial reimbursement | 4 | Bartlett 2018,34 Harth 1990,51 Skinner 2011,85 Taylor 2015a,92 | |
Trust | Trust in clinician | 13 | Chantler 2007,38 Chappuy 2006,39 Dahan 2020,22 Dreyzin 2014,43 Eiser 2005,44 Greenberg 2017,50 Ingersgaard 2018,60 Jollye 2009,63 Mwangi 2017,75 Nieminen 2015a,76 Pagano-Therrien 2017,78 Snowdon 2006,86 Woodgate 2010.97 |
Trust in medical researchers | 4 | Helgesson 2009,54 Jenkins 2009,62 Shah 2018a,23 Snowdon 2006.86 | |
Trust in particular institution | 3 | Harth 1990,51 Jenkins 2009,62 Vecchi 2013.94 | |
Trust in medicine in general | 2 | Clausen 1954,40 Tait 2004.90 | |
Trust in research | 4 | Chantler 2007,38 Clausen 1954,40 Hoberman 2013a,55 Paquette 2019.80 | |
Trust (other) | 3 | Chantler 2007a,38 Jenkins 2009,62 Tait 2003a.89 | |
Support of research | Altruism | 30 | Barakat 2014,33 Chantler 2007,38 D'Amanda 2019,41 Dahan 2020,22 Eiser 2005,44 Harth 1990,51 Harth 1992,52 Hoberman 2013a,55 Hoehn 2005,56 Ingersgaard 2018,60 Jenkins 2009,62 Jollye 2009,63 Langley 1998,68 Mason 2000,69 Morley 2005,74 Nieminen 2015a,76 Pagano-Therrien 2017,78 Paquette 2019,80 Read 2009,81 Sammons 2007,82 Shah 2018a,23 Shilling 2009,84 Tait 1998,91 Tait 2003a,89 Thomas 2013,93 Vecchi 2013,94 Ward 2009,96 Woodgate 2010,97 Woolfall 2013,98 Zupancic 1997a.24 |
Importance of medical research | 26 | Bartlett 2018,34 Buck 2015,37 Chantler 2007,38 Clausen 1954,40 Dahan 2020,22 Dreyzin 2014,43 Harth 1990,51 Harth 1992,52 Hayman 2001,53 Helgesson 2009,54 Hoberman 2013a,55 Jenkins 2009,62 Jollye 2009,63 Kumari 2019,66 Langley 1998,68 Pagano-Therrien 2017,78 Sammons 2007,82 Shah 2018a,23 Shilling 2009,84 Skinner 2011,85 Snowdon 2006,86 Tait 1998,91 Tait 2003a,89 Thomas 2013,89 Vecchi 2013,94 Wynn 2010.99 | |
Informational and consent process related | Felt as only option | 7 | Chappuy 2006,39 Dahan 2020,22 Hayman 2001,53 Snowdon 2006,86 Ward 2009,96 Woodgate 2010,97 Zupancic 1997.24 |
Understanding of research materials | 2 | Tait 1998,91 Tait 2003a.89 | |
Experience or lack of experience with research | 8 | Baxter 2012a,35 Chantler 2007,38 Chappuy 2006a,39 Jay 2007a,61 Pagano-Therrien 2017,78 Read 2009a,81 Tait 2003a,89 Zupancic 1997a.24 | |
Relational | Positive relationship to researcher | 15 | Bartlett 2018,34 Dahan 2020,22 Harth 1990,51 Hoberman 2013a,55 Jay 2007a,61 Pagano-Therrien 2017,78 Snowdon 2006,86 Sureshkumar 2012a,87 Tait 1998,91 Tait 2003a,89 Tait 2004,90 Thomas 2013,93 Ward 2009,96 Woodgate 2010,97 Woolfall 2013.98 |
Influence of family and friends | 11 | Bartlett 2018,34 Baxter 2012,35 Clausen 1954a,40 Harth 1990,51 Helgesson 2009,54 Hoberman 2013a,55 Jollye 2009,63 Read 2009,81 Skinner 2011,85 Tait 2003a,89 Woodgate 2010.97 | |
Participation encouraged by clinical team | 11 | Clausen 1954a,40 Harth 1990,51 Langley 1998,68 Mazzocco 1999a,70 Paquette 2019,80 Read 2009,81 Sammons 2007,82 Snowdon 2006,86 Woodgate 2010,97 Woolfall 2013,98 Zupancic 1997a.24 | |
Child’s desire to participate | 2 | Pagano-Therrien 2017,78 Paquette 2019.80 |
Category . | Facilitators . | Number of Studies . | Individual Studies . |
---|---|---|---|
Benefits | Health benefit to child | 39 | Barakat 2014,33 Buck 2015,37 Chantler 2007,38 Chappuy 2006,39 Clausen 1954a,40 D'Amanda 2019,a,41 Dahan 2020,22 Dreyzin 2014a,43 Eiser 2005,44 Greenberg 2017,50 Harth 1990,51 Harth 1992,52 Hayman 2001,53 Helgesson 2009,54 Hoberman 2013a,55 Hoehn 2005,56 Ingersgaard 2018,60 Jollye 2009,63 Kumari 2019,66 Langley 1998,68 Mason 2000,69 Morley 2005,74 Mwangi 2017a,75 Nieminen 2015,76 Paquette 2019,80 Read 2009,81 Sammons 2007,82 Shah 2018a,23 Shilling 2009,84 Snowdon 2006,86 Tait 1998,91 Tait 2003a,89 Tait 2004a,90 Thomas 2013,93 Ward 2009,96 Woodgate 2010,97 Woolfall 2013,98 Wynn 2010,99 Zupancic 1997a,24 |
Nonhealth benefit to child | 8 | Chantler 2007,38 D'Amanda 2019,41 Helgesson 2009,54 Shilling 2009,84 Skinner 2011,85 Tait 1998,91 Thomas 2013,93 Wynn 2010.99 | |
More contact with medical team | 5 | Chantler 2007,38 Dreyzin 2014,43 Harth 1990,52 Mwangi 2017a.75 | |
Direct benefit to parent | 6 | Chantler 2007,38 Harth 1990,51 Helgesson 2009,54 Langley 1998,68 Skinner 2011,85 Ward 2009.96 | |
Minimal burden to child or parent | 16 | Buck 2015,37 Dreyzin 2014,43 Eiser 2005,44 Greenberg 2017,50 Helgesson 2009,54 Hoberman 2013a,55 Mason 2000,69 Pagano-Therrien 2017,78 Paquette 2019,80 Read 2009,81 Sammons 2007,82 Shilling 2009,84 Skinner 2011,85 Taylor 2015a,92 Tait 1998,91 Woodgate 2010.97 | |
Financial reimbursement | 4 | Bartlett 2018,34 Harth 1990,51 Skinner 2011,85 Taylor 2015a,92 | |
Trust | Trust in clinician | 13 | Chantler 2007,38 Chappuy 2006,39 Dahan 2020,22 Dreyzin 2014,43 Eiser 2005,44 Greenberg 2017,50 Ingersgaard 2018,60 Jollye 2009,63 Mwangi 2017,75 Nieminen 2015a,76 Pagano-Therrien 2017,78 Snowdon 2006,86 Woodgate 2010.97 |
Trust in medical researchers | 4 | Helgesson 2009,54 Jenkins 2009,62 Shah 2018a,23 Snowdon 2006.86 | |
Trust in particular institution | 3 | Harth 1990,51 Jenkins 2009,62 Vecchi 2013.94 | |
Trust in medicine in general | 2 | Clausen 1954,40 Tait 2004.90 | |
Trust in research | 4 | Chantler 2007,38 Clausen 1954,40 Hoberman 2013a,55 Paquette 2019.80 | |
Trust (other) | 3 | Chantler 2007a,38 Jenkins 2009,62 Tait 2003a.89 | |
Support of research | Altruism | 30 | Barakat 2014,33 Chantler 2007,38 D'Amanda 2019,41 Dahan 2020,22 Eiser 2005,44 Harth 1990,51 Harth 1992,52 Hoberman 2013a,55 Hoehn 2005,56 Ingersgaard 2018,60 Jenkins 2009,62 Jollye 2009,63 Langley 1998,68 Mason 2000,69 Morley 2005,74 Nieminen 2015a,76 Pagano-Therrien 2017,78 Paquette 2019,80 Read 2009,81 Sammons 2007,82 Shah 2018a,23 Shilling 2009,84 Tait 1998,91 Tait 2003a,89 Thomas 2013,93 Vecchi 2013,94 Ward 2009,96 Woodgate 2010,97 Woolfall 2013,98 Zupancic 1997a.24 |
Importance of medical research | 26 | Bartlett 2018,34 Buck 2015,37 Chantler 2007,38 Clausen 1954,40 Dahan 2020,22 Dreyzin 2014,43 Harth 1990,51 Harth 1992,52 Hayman 2001,53 Helgesson 2009,54 Hoberman 2013a,55 Jenkins 2009,62 Jollye 2009,63 Kumari 2019,66 Langley 1998,68 Pagano-Therrien 2017,78 Sammons 2007,82 Shah 2018a,23 Shilling 2009,84 Skinner 2011,85 Snowdon 2006,86 Tait 1998,91 Tait 2003a,89 Thomas 2013,89 Vecchi 2013,94 Wynn 2010.99 | |
Informational and consent process related | Felt as only option | 7 | Chappuy 2006,39 Dahan 2020,22 Hayman 2001,53 Snowdon 2006,86 Ward 2009,96 Woodgate 2010,97 Zupancic 1997.24 |
Understanding of research materials | 2 | Tait 1998,91 Tait 2003a.89 | |
Experience or lack of experience with research | 8 | Baxter 2012a,35 Chantler 2007,38 Chappuy 2006a,39 Jay 2007a,61 Pagano-Therrien 2017,78 Read 2009a,81 Tait 2003a,89 Zupancic 1997a.24 | |
Relational | Positive relationship to researcher | 15 | Bartlett 2018,34 Dahan 2020,22 Harth 1990,51 Hoberman 2013a,55 Jay 2007a,61 Pagano-Therrien 2017,78 Snowdon 2006,86 Sureshkumar 2012a,87 Tait 1998,91 Tait 2003a,89 Tait 2004,90 Thomas 2013,93 Ward 2009,96 Woodgate 2010,97 Woolfall 2013.98 |
Influence of family and friends | 11 | Bartlett 2018,34 Baxter 2012,35 Clausen 1954a,40 Harth 1990,51 Helgesson 2009,54 Hoberman 2013a,55 Jollye 2009,63 Read 2009,81 Skinner 2011,85 Tait 2003a,89 Woodgate 2010.97 | |
Participation encouraged by clinical team | 11 | Clausen 1954a,40 Harth 1990,51 Langley 1998,68 Mazzocco 1999a,70 Paquette 2019,80 Read 2009,81 Sammons 2007,82 Snowdon 2006,86 Woodgate 2010,97 Woolfall 2013,98 Zupancic 1997a.24 | |
Child’s desire to participate | 2 | Pagano-Therrien 2017,78 Paquette 2019.80 |
Study directly compared by enrollment status. See Table 7 for more detail.
Comparison of Facilitators for Participation in Pediatric Research between Participants and Nonparticipants
Category . | Facilitators Directly Compared . | Study . | Discussion . | Statistics . |
---|---|---|---|---|
Benefits | Health benefit to child | Clausen 1954b,40 | More consenting mothers (66%) than decliners (7%) reported believing that the vaccine had the potential to prevent polio | NA |
D’Amanda 2019b,41 | More joiners (8/16) than decliners (2/15) believed medication usage would have a positive effect on health | NA | ||
Dreyzin 2014b,43 | More consenters (5/5) than decliners (0/1) reported being motivated by “an acute deterioration in their child’s health” | NA | ||
Hoberman 2013a,55 | Consenters had higher “benefit to my child” scores than nonconsenters | P < .001 | ||
a | Consenters were more likely than nonconsenters to endorse that the “study may enhance standard of care” | P < .001 | ||
Mwangi 2017b,75 | More participants (13/20) than decliners (1/15) discussed “potential therapeutic benefit to child of participating” | NA | ||
bMore participants (18/20) than decliners (3/15) “demonstrated belief that participants got malaria treatment” | NA | |||
Shah 2018a,23 | Enrollers (101/105) were more likely than decliners (41/58) to rate “the potential benefit to my child” as “very important or important” to their decision making | P < .001 | ||
Tait 2003a,89 | Consenters had higher scores on “perceived study as benefit to child” compared with nonconsenters | P < .001 | ||
Tait 2004a,90 | Consenters were more likely than decliners to view the risk-benefit ratio as lower | P < .001 | ||
More contact with medical team | Zupancic 1997a,24 | Consenters assessed study as of greater benefit and lower risk compared with nonconsenters | P < .0001 | |
Mwangi 2017c,75 | Participants (12/20) and decliners (11/15) “discussed a general perception of benefit in access to ancillary clinical services for participants” | NA | ||
cParticipants (6/20) and decliners (7/15) “demonstrated belief that there was a significant benefit in access to ancillary clinical services for participants” | NA | |||
Minimal burden to child | Hoberman 2013a,55 | Consenters were more likely than nonconsenters to perceive the study as low risk | P < .001 | |
Taylor 2015e,92 | Patients in non-brief studies were more likely to participate than those in brief studies | OR 1.7, P < .001 | ||
a | Patients in noninvasive studies were more likely to participant than those in invasive studies | OR 2.9, P < .001 | ||
Financial reimbursement | Taylor 2015e,92 | Patients in studies with no compensation were more likely to participate than those with compensation | OR 1.9, P < .001 | |
Trust | Trust in clinician | Nieminen 2015a,76 | Consenters (771/956) were more likely than nonconsenters (n = 242/356) to agree or strongly agree with the statement: “I trust my own well-baby-clinic nurse in matters related to my child's health” | P < .001 |
Trust in medical researchers | Shah 2018a,23 | Enrollers (87%) were more likely than decliners (63%) to report trusting the person who discussed the PENUT trial with them | P < .001 | |
Trust in research | Hoberman 2013a,55 | Consenters had higher “trust in research” scores than nonconsenters | P < .001 | |
Chantler 2007b,38 | More parents who enrolled than parents who declined enrollment reported a general trust in vaccines | NA | ||
Trust (other) | Tait 2003a,89 | Consenters had higher “trust” scores (12.1 on scale of 3–15) than nonconsenters (11.1) | P < .001 | |
Support of research | Altruism | Hoberman 2013a,55 | Consenters had higher altruism scores than nonconsenters | P < .001 |
Nieminen 2015b,76 | More consenters (742/965) than nonconsenters (241/356) reported that “promoting the common good” had an impact on their enrollment decision | NA | ||
Shah 2018a,23 | Enrollers (96/105) were more likely than decliners (37/58) to rate “the potential benefit to future premature babies” as “very important or important” | P < .001 | ||
Tait 2003a,89 | Consenters had higher perception of the “benefit to others” (7.8 on a scale of 1–10) than nonconsenters (5.9) | P < .001 | ||
c | Consenters had no difference in altruism construct score (12.9 on scale of 1–15) than nonconsenters (12.7) | P = NS | ||
Zupancic 1997b,24 | More consenters (98%) than decliners (84%) considered “altruistic motives” as important in their decision | NA | ||
Importance of medical research | Hoberman 2013a,55 | Consenters had higher “importance of research study” scores than nonconsenters | P < .001 | |
Shah 2018a,23 | Enrollers (86/105) were more likely than decliners (23/58) to rate “the contribution to science and medicine” as “very important or important” | P < .001 | ||
Tait 2003a89 | Consenters viewed the study as more important (8.0 on scale of 1–10) than nonconsenters (5.9) | P < .001 | ||
Informational and consent process related | Understanding of research materials | Tait 2003a89 | Consenters were more likely to have listened completely (89.3%) than nonconsenters (67.8%) | P < .0001 |
a | Consenters were more likely to have read the consent completely (62.0%) than nonconsenters (43.0%) | P < .0001 | ||
a | Consenters were more likely to have completely understood the consent (77.5%) than nonconsenters (58.4%) | P < .0001 | ||
Experience or lack of experience with research | Baxter 2012c,89 | Parents were not more likely to enroll their child if they had another child participating in the same study | P = NS | |
Chappuy 2006c,39 | Those who enrolled and those who declined did not differ in prior experience with research | P = NS | ||
Jay 2007a,61 | Consenters to a follow up study were more likely than non-consenters to report a positive recollection of their child's care at past study visits | P = .001 | ||
Read 2009a,81 | Parents who had participated in research themselves were more likely to authorize their child to participate in a research trial | P < .001 | ||
Tait 2003c,89 | Consenters did not differ in prior research experience of the child (20.0%) compared with nonconsenters (18.3%) | P = NS | ||
c | Consenters did not differ in prior research experience of the parent (24.4%) compared with nonconsenters (21.5%) | P = NS | ||
Zupancic 1997c,24 | Consenters (17%) and nonconsenters (22%) reported similar rates of previous research involvement | NA | ||
Relational | Positive relationship to researcher | Hoberman 2013a,55 | Consenters had higher scores for “perceived friendliness and professionalism of researcher” than nonconsenters | P < .003 |
Jay 2007a,61 | “Study staff contactable at any time to answer query” influenced parents to consent | P = .01 | ||
Sureshkumar 2012a,87 | Referral to the study by a pediatrician or nephrologist increased likelihood of consent compared with referral from emergency department | P < .0001 | ||
Recruiting physician being part of the research team increased likelihood of consent | RR 1.9, P < .001, | |||
Tait 2003a,89 | Consenters were more likely (97%) than nonconsenters (87%) to view researcher as “friendly and relaxed” | P < .0001 | ||
Influence of family and friends | Clausen 1954b,40 | More consenters (61%) than decliners (39%) discussed with family and friends | NA | |
Hoberman 2013c,55 | Consenters did not differ from nonconsenters in responses to question set on “external influences” | P = NS | ||
Tait 2003c,89 | Consenters (52.0%) and nonconsenters (55.9%) did not differ in “discussed the study information with others before decision” | P = NS | ||
c | Consenters (40.3%) and nonconsenters (46.3%) did not differ in “discussed with spouse” | P = NS | ||
Participation encouraged by clinical team | Clausen 1954b,40 | More consenters (41%) than decliners (27%) discussed study with doctor or nurse before making decision about vaccine | NA | |
Mazzocco 1999a,70 | Enrollment was higher when the research was introduced by a physician (73.3%) than by research staff (58.7%) | P < .01 | ||
Zupancic 1997c,24 | Some consenters (34/103) and nonconsenters (11/37) both endorsed that they “prefer to have the doctor advise me whether the baby should be in the study, rather than asking me to decide” | NA |
Category . | Facilitators Directly Compared . | Study . | Discussion . | Statistics . |
---|---|---|---|---|
Benefits | Health benefit to child | Clausen 1954b,40 | More consenting mothers (66%) than decliners (7%) reported believing that the vaccine had the potential to prevent polio | NA |
D’Amanda 2019b,41 | More joiners (8/16) than decliners (2/15) believed medication usage would have a positive effect on health | NA | ||
Dreyzin 2014b,43 | More consenters (5/5) than decliners (0/1) reported being motivated by “an acute deterioration in their child’s health” | NA | ||
Hoberman 2013a,55 | Consenters had higher “benefit to my child” scores than nonconsenters | P < .001 | ||
a | Consenters were more likely than nonconsenters to endorse that the “study may enhance standard of care” | P < .001 | ||
Mwangi 2017b,75 | More participants (13/20) than decliners (1/15) discussed “potential therapeutic benefit to child of participating” | NA | ||
bMore participants (18/20) than decliners (3/15) “demonstrated belief that participants got malaria treatment” | NA | |||
Shah 2018a,23 | Enrollers (101/105) were more likely than decliners (41/58) to rate “the potential benefit to my child” as “very important or important” to their decision making | P < .001 | ||
Tait 2003a,89 | Consenters had higher scores on “perceived study as benefit to child” compared with nonconsenters | P < .001 | ||
Tait 2004a,90 | Consenters were more likely than decliners to view the risk-benefit ratio as lower | P < .001 | ||
More contact with medical team | Zupancic 1997a,24 | Consenters assessed study as of greater benefit and lower risk compared with nonconsenters | P < .0001 | |
Mwangi 2017c,75 | Participants (12/20) and decliners (11/15) “discussed a general perception of benefit in access to ancillary clinical services for participants” | NA | ||
cParticipants (6/20) and decliners (7/15) “demonstrated belief that there was a significant benefit in access to ancillary clinical services for participants” | NA | |||
Minimal burden to child | Hoberman 2013a,55 | Consenters were more likely than nonconsenters to perceive the study as low risk | P < .001 | |
Taylor 2015e,92 | Patients in non-brief studies were more likely to participate than those in brief studies | OR 1.7, P < .001 | ||
a | Patients in noninvasive studies were more likely to participant than those in invasive studies | OR 2.9, P < .001 | ||
Financial reimbursement | Taylor 2015e,92 | Patients in studies with no compensation were more likely to participate than those with compensation | OR 1.9, P < .001 | |
Trust | Trust in clinician | Nieminen 2015a,76 | Consenters (771/956) were more likely than nonconsenters (n = 242/356) to agree or strongly agree with the statement: “I trust my own well-baby-clinic nurse in matters related to my child's health” | P < .001 |
Trust in medical researchers | Shah 2018a,23 | Enrollers (87%) were more likely than decliners (63%) to report trusting the person who discussed the PENUT trial with them | P < .001 | |
Trust in research | Hoberman 2013a,55 | Consenters had higher “trust in research” scores than nonconsenters | P < .001 | |
Chantler 2007b,38 | More parents who enrolled than parents who declined enrollment reported a general trust in vaccines | NA | ||
Trust (other) | Tait 2003a,89 | Consenters had higher “trust” scores (12.1 on scale of 3–15) than nonconsenters (11.1) | P < .001 | |
Support of research | Altruism | Hoberman 2013a,55 | Consenters had higher altruism scores than nonconsenters | P < .001 |
Nieminen 2015b,76 | More consenters (742/965) than nonconsenters (241/356) reported that “promoting the common good” had an impact on their enrollment decision | NA | ||
Shah 2018a,23 | Enrollers (96/105) were more likely than decliners (37/58) to rate “the potential benefit to future premature babies” as “very important or important” | P < .001 | ||
Tait 2003a,89 | Consenters had higher perception of the “benefit to others” (7.8 on a scale of 1–10) than nonconsenters (5.9) | P < .001 | ||
c | Consenters had no difference in altruism construct score (12.9 on scale of 1–15) than nonconsenters (12.7) | P = NS | ||
Zupancic 1997b,24 | More consenters (98%) than decliners (84%) considered “altruistic motives” as important in their decision | NA | ||
Importance of medical research | Hoberman 2013a,55 | Consenters had higher “importance of research study” scores than nonconsenters | P < .001 | |
Shah 2018a,23 | Enrollers (86/105) were more likely than decliners (23/58) to rate “the contribution to science and medicine” as “very important or important” | P < .001 | ||
Tait 2003a89 | Consenters viewed the study as more important (8.0 on scale of 1–10) than nonconsenters (5.9) | P < .001 | ||
Informational and consent process related | Understanding of research materials | Tait 2003a89 | Consenters were more likely to have listened completely (89.3%) than nonconsenters (67.8%) | P < .0001 |
a | Consenters were more likely to have read the consent completely (62.0%) than nonconsenters (43.0%) | P < .0001 | ||
a | Consenters were more likely to have completely understood the consent (77.5%) than nonconsenters (58.4%) | P < .0001 | ||
Experience or lack of experience with research | Baxter 2012c,89 | Parents were not more likely to enroll their child if they had another child participating in the same study | P = NS | |
Chappuy 2006c,39 | Those who enrolled and those who declined did not differ in prior experience with research | P = NS | ||
Jay 2007a,61 | Consenters to a follow up study were more likely than non-consenters to report a positive recollection of their child's care at past study visits | P = .001 | ||
Read 2009a,81 | Parents who had participated in research themselves were more likely to authorize their child to participate in a research trial | P < .001 | ||
Tait 2003c,89 | Consenters did not differ in prior research experience of the child (20.0%) compared with nonconsenters (18.3%) | P = NS | ||
c | Consenters did not differ in prior research experience of the parent (24.4%) compared with nonconsenters (21.5%) | P = NS | ||
Zupancic 1997c,24 | Consenters (17%) and nonconsenters (22%) reported similar rates of previous research involvement | NA | ||
Relational | Positive relationship to researcher | Hoberman 2013a,55 | Consenters had higher scores for “perceived friendliness and professionalism of researcher” than nonconsenters | P < .003 |
Jay 2007a,61 | “Study staff contactable at any time to answer query” influenced parents to consent | P = .01 | ||
Sureshkumar 2012a,87 | Referral to the study by a pediatrician or nephrologist increased likelihood of consent compared with referral from emergency department | P < .0001 | ||
Recruiting physician being part of the research team increased likelihood of consent | RR 1.9, P < .001, | |||
Tait 2003a,89 | Consenters were more likely (97%) than nonconsenters (87%) to view researcher as “friendly and relaxed” | P < .0001 | ||
Influence of family and friends | Clausen 1954b,40 | More consenters (61%) than decliners (39%) discussed with family and friends | NA | |
Hoberman 2013c,55 | Consenters did not differ from nonconsenters in responses to question set on “external influences” | P = NS | ||
Tait 2003c,89 | Consenters (52.0%) and nonconsenters (55.9%) did not differ in “discussed the study information with others before decision” | P = NS | ||
c | Consenters (40.3%) and nonconsenters (46.3%) did not differ in “discussed with spouse” | P = NS | ||
Participation encouraged by clinical team | Clausen 1954b,40 | More consenters (41%) than decliners (27%) discussed study with doctor or nurse before making decision about vaccine | NA | |
Mazzocco 1999a,70 | Enrollment was higher when the research was introduced by a physician (73.3%) than by research staff (58.7%) | P < .01 | ||
Zupancic 1997c,24 | Some consenters (34/103) and nonconsenters (11/37) both endorsed that they “prefer to have the doctor advise me whether the baby should be in the study, rather than asking me to decide” | NA |
NS, not significant; OR, odds ratio; RR, risk ratio.
Significantly associated with enrolling (quantitative only: P < .05).
Tends toward enrolling (quantitative: [P ≥ .05 and P ≤ .1] or qualitative).
No difference (quantitative or qualitative).
Tends toward declining (quantitative: [P ≥ .05 and P ≤ .1] or qualitative).
Significantly associated with declining (quantitative only: P < .05).
Benefits
The perceived benefits of pediatric research participation were strong facilitators for research participation. The most frequently reported facilitator was health benefit to child (N = 39) with nearly all studies that compared by enrollment status reporting that the perception of health benefit to child was associated with enrollment. A belief that participation was a minimal burden to child or parent (N = 16) was also associated with enrollment. Four studies identified financial reimbursement as a facilitator to research participation. The single manuscript that compared by enrollment status looked at research participation in a pediatric emergency department and found that studies without compensation had higher enrollment for moderate to very invasive studies.92 Because invasive studies were more likely to have compensation, the authors were unable to compare similar studies with and without compensation to fully understand the effects of compensation.
Trust
Trust emerged as an important theme in pediatric research participation. Studies frequently reported trust as a facilitator to pediatric research participation including trust in clinicians (N = 13), medical researchers (N = 4), medical institutions (N = 3), medicine (N = 2), medical research (N = 4), as well as undefined trust (N = 1), trust in vaccines (N = 1) and trust in government (N = 1). For each trust category, all studies that compared by enrollment status (N = 5) found greater trust to be associated with enrollment.
Support of Research
Another major theme was parental support of research. Within this category, we included altruism, importance of medical research, and prior experience with research. Studies found altruism (N = 30) to be the second most frequently reported facilitator surpassed only by health benefit to child (N = 39). Studies also reported the importance of research (N = 26) as a facilitator to research participation. For each of these 3 categories, all studies that compared parents by enrollment status found an association between the facilitator and enrollment.
Informational and Consent Process Related
Facilitators emerged related to the consent process and information sharing. These included research participation being “felt as the only option,” understanding of research materials, and prior experience with research. All studies that compared by enrollment status found greater understanding among those who enrolled. Prior research experience was variably associated with enrollment status.
Relational
Several facilitators to participation emerged related to parental relationships. These included positive relationship to researcher, influence of families and friends, clinical team encouragement of participation, and a child’s desire to participate. The most frequently reported facilitators in this category were a positive relationship with researcher (N = 15), influence of family and friends (N = 11), and clinical team encouragement of participation (N = 11). For clinical team encouragement, all studies that compared by enrollment status found an association between this facilitator and enrollment.
Changes Over Time
Facilitators to research were examined across decades to assess for changes over time. Nearly all included studies (N = 67 of 70) were published in the 1990s (N = 6), 2000s (N = 22), or 2010s (N = 39). The most cited facilitators to enrollment in each of these 3 epochs (1990s, 2000s, 2010s) were health benefit to child (N = 5, N = 15, N = 17), altruism (N = 5, N = 11, N = 13), and importance of research (N = 4, N = 8, N = 12). No trends suggesting changes over time were evident.
Qualitative Meta-summary of Barriers
Tables 8 and 9 show the barriers to pediatric research. In Table 8, we share the number and name of each study that included the barrier. In Table 9, we present only studies that directly compared the barrier between enrolled and declined parents. For quantitative comparisons, we present the relevant statistic of the differences by enrollment status.
Barriers for Participation in Pediatric Research
Category . | Barriers . | Number of Studies . | Individual Studies . |
---|---|---|---|
Concerns about study participation | Risk to child | 46 | Bartlett 2018,34 Braga 2014,36 Buck 2015,37 Chantler 2007,38 Chappuy 2006,39 Clausen 1954a,40 D'Amanda 2019a,41 Dreyzin 2014,43 Eiser 2005,44 Elemraid 2013,45 Gill 2014,48 Greenberg 2017,50 Harth 1990,51 Hayman 2001a,53 Helgesson 2009,54 Hoberman 2013a,55 Hoehn 2005,56 Howard Sharp 2020,58 Hulst 2005,59 Ingersgaard 2018,60 Jollye 2009,63 Korotchikova 2010,65 Kumari 2019,66 Langley 1998,68 Mason 2000,69 Menon 2012,71 Menon 2014,72 Mwangi 2017a,75 Nieminen 2015,76 Norris 2010,77 Papaz 2012,79 Paquette 2019,80 Read 2009,81 Shah 2018a,23 Shilling 2009,84 Snowdon 2006,86 Surun 2018,88 Tait 1998,91 Tait 2003a,89 Tait 2004a,90 Thomas 2013,93 Volkening 2017,95 Ward 2009,96 Woodgate 2010,97 Woolfall 2013,98 Zupancic 1997a.24 |
Negative impact on medical care | 21 | Barakat 2014,33 Baxter 2012,35 Braga 2014,36 Buck 2015,37 Clausen 1954,40 D'Amanda 2019,41 Elemraid 2013,45 Gattuso 2006,46 Genetti 2019,47 Gonzalez 2016,49 Helgesson 2009,54 Hoberman 2013a,55 Ingersgaard 2018,60 Menon 2014,72 Mihrshahi 2002,73 Morley 2005,74 Sureshkumar 2012,87 Tait 1998,91 Tait 2003a,89 Taylor 2015,92 Woolfall 2013.98 | |
Lack of direct benefit to child | 16 | Barakat 2014,33 Bartlett 2018,34 Clausen 1954,40 D'Amanda 2019a,41 Gattuso 2006,46 Genetti 2011,47 Gill 2014,48 Greenberg 2017,50 Helgesson 2009,54 Howard Sharp 2020,58 Kick 2018,64 Menon 2014,72 Mihrshahi 2002,73 Read 2009,81 Skinner 2011,85 Sureshkumar 2012.87 | |
Child's own medical concerns | 9 | Chappuy 2006,39 Hulst 2005,59 Menon 2014,72 Mihrshahi 2002,73 Morley 2005,74 Sureshkumar 2012,87 Taylor 2015,92 Thomas 2013,93 Woodgate 2010.97 | |
Phlebotomy concerns | 14 | Chantler 2007,38 D'Amanda 2019a,41 Dlugos 2005,42 Helgesson 2009,54 Jay 2007a,61 Jenkins 2009,62 Kick 2018,64 Labib 2018,67 Mazzocco 1999,70 Menon 2012,71 Menon 2014,72 Scollon 2014,83 Vecchi 2013,94 Wynn 2010.99 | |
Burdens | Logistics or burden of participation | 35 | Bartlett 2018,34 Baxter 2012,35 Chappuy 2006,39 D'Amanda 2019a,41 Gattuso 2006,46 Genetti 2019,47 Gill 2014,48 Gonzalez 2016,49 Greenberg 2017,50 Harth 1990,51 Hayman 2001,53 Helgesson 2009,54 Hoehn 2009,56 Hulst 2005,59 Jenkins 2009,62 Kick 2018,64 Korotchikova 2010,65 Langley 1998,68 Mason 2000,69 Mazzocco 1999,70 Mihrshahi 2002,73 Papaz 2012,79 Paquette 2019,80 Read 2009,81 Shilling 2009,84 Skinner 2011,85 Sureshkumar 2012,87 Tait 1998,91 Tait 2003a,89 Taylor 2015,92 Vecchi 2013,94 Volkening 2017,95 Woolfall 2013,98 Wynn 2010,99 Zupancic 1997a.24 |
Lack of trust | Lack of trust in clinicians | 1 | Chappuy 2006.39 |
Lack of trust in medical researchers | 6 | Greenberg 2017,50 Helgesson 2009,54 Mason 2000,69 Read 2009,81 Shah 2018a,23 Wynn 2010.99 | |
Lack of trust in particular institution | 2 | Harth 1990,51 Morley 2005.74 | |
Lack of trust in medicine in general | 2 | Buck 2015,37 Harth 1990.51 | |
Lack of trust in research | 4 | Chantler 2007,38 Dreyzin 2014,43 Mazzocco 1999,70 Snowdon 2006.86 | |
General research concerns | Lack of interest in research | 18 | Braga 2014,36 Elemraid 2013,45 Gattuso 2006,46 Genetti 2019,47 Gonzalez 2016,49 Harth 1990,51 Hulst 2005,59 Langley 1998,68 Mazzocco 1999,70 Menon 2012,71 Menon 2014,72 Mihrshahi 2002,73 Morley 2005,74 Papaz 2012,79 Sammons 2007,82 Skinner 2011,85 Taylor 2015,92 Volkening 2017.95 |
Guinea pig concerns | 8 | Buck 2015,37 Chantler 2007,38 Clausen 1954,40 Greenberg 2017,50 Hoehn 2005,56 Korotchikova 2010,65 Shah 2018a,23 Tait 1998.91 | |
Privacy concerns | 12 | Gattuso 2006,46 Genetti 2019,47 Gill 2014,48 Harth 1990,51 Howard Sharp 2020,58 Kick 2018,64 Kumari 2019,66 Menon 2012,71 Papaz 2012,79 Sammons 2007,82 Scollon 2014,83 Tait 2004.90 | |
Lack of belief in research | 6 | Helgesson 2009,44 Hulst 2005,59 Labib 2018,67 Nieminen 2015,76 Taylor 2015,92 Tait 1998a.91 | |
Discomfort with randomization | 18 | Braga 2014,54 Buck 2015,37 Eiser 2005,76 Gonzalez 2016,49 Greenberg 2017,50 Hoberman 2013a,55 Ingersgaard 2018,60 Jollye 2009,63 Kick 2018,64 Nieminen 2015,76 Norris 2010,77 Sammons 2007,82 Snowdon 2006,86 Surun 2018,88 Tait 1998,91 Woodgate 2010,97 Woolfall 2013,98 Wynn 2010.99 | |
Genetic testing concerns | 5 | Genetti 2019,47 Howard Sharp 2020,58 Papaz 2012,79 Scollon 2014,83 Skinner 2011.85 | |
Informational and consent process related | Not enough information provided about study; inadequate understanding of study | 16 | Barakat 2014,33 Chappuy 2006,39 Dahan 2020,22 Dreyzin 2014,43 Eiser 2005,44 Gill 2014,48 Helgesson 2009,54 Hoberman 2013a,55 Korotchikova 2010,65 Menon 2012,71 Menon 2014,72 Mwangi 2017a,75 Paquette 2019,80 Read 2009,81 Tait 2003a,89 Tait 2004.90 |
Not enough time to make decision about enrollment | 13 | Barakat 2014,33 Chappuy 2006,39 Eiser 2005,44 Hoehn 2009a,56 Jollye 2009,63 Menon 2012,71 Papaz 2012,79 Paquette 2019,80 Snowdon 2006,86 Tait 1998,91 Tait 2004,90 Thomas 2013,93 Ward 2009.96 | |
Not enough knowledge about medical condition or lack of comfort with clinical decision | 2 | Mazzocco 1999,70 Read 2009a.81 | |
Decision too stressful | 29 | Dahan 2020,22 Dlugos 2005,42 Genetti 2019,47 Greenberg 2017,50 Hoberman 2013a,55 Howard Sharp 2020,58 Hulst 2005,59 Ingersgaard 2018,60 Jollye 2009,63 Labib 2018,67 Mason 2000,69 Menon 2012,71 Menon 2014,72 Norris 2010,77 Papaz 2012,79 Paquette 2019,80 Read 2009,81 Sammons 2007,82 Scollon 2014,83 Shah 2018a,23 Snowdon 2006,86 Tait 1998a,91 Tait 2003a,90 Tait 2004,89 Thomas 2013,93 Ward 2009,96 Woodgate 2010,97 Woolfall 2013,98 Zupancic 1997a.24 | |
Parental perception of illness | 10 | Barakat 2014,33 Bartlett 2018,34 Buck 2015,37 Chantler 2007a,38 Clausen 1954,40 D'Amanda 2019a,41 Dreyzin 2014a,43 Jollye 2009,63 Wynn 2010,99 Zupancic 1997a.24 | |
Language barrier | 2 | Helgesson 2009,54 Papaz 2012.79 | |
Prior experience or lack of experience with research | 13 | Chantler 2007a,38 Dahan 2020,22 Gonzalez 2016,49 Hulst 2005,59 Menon 2012,71 Menon 2014,72 Nieminen 2015a,76 Paquette 2019a,80 Shah 2018a,23 Tait 1998a,91 Tait 2003a,90 Tait 2004,89 Thomas 2013a.93 | |
Relational | Discomfort with proxy nature of consent | 3 | Chantler 2007,38 Papaz 2012,79 Woodgate 2010.97 |
One parent wanted to enroll, other did not | 8 | Dahan 2020,22 Genetti 2019,47 Hoehn 2009,56 Jenkins 2009,62 Korotchikova 2010a,65 Langley 1998,68 Menon 2014,72 Skinner 2011.85 | |
Family problems | 5 | Baxter 2012,35 Chantler 2007,38 Jay 2007a,61 Menon 2012,71 Mihrshahi 2002.73 | |
Feeling pressured | 2 | Hayman 2001a,53 Mason 2000.69 | |
Child's wishes related to enrollment | 15 | Chantler 2007,38 Chappuy 2006,39 Clausen 1954,40 Gill 2014,48 Greenberg 2017,50 Howard Sharp 2020,58 Hulst 2005,59 Ingersgaard 2018,60 Jay 2007a,61 Menon 2014,72 Shilling 2009,84 Sureshkumar 2012,87 Tait 2003a,90 Woodgate 2010,97 Woolfall 2013.98 | |
Decision-making style | 1 | Hoberman 2013a55 |
Category . | Barriers . | Number of Studies . | Individual Studies . |
---|---|---|---|
Concerns about study participation | Risk to child | 46 | Bartlett 2018,34 Braga 2014,36 Buck 2015,37 Chantler 2007,38 Chappuy 2006,39 Clausen 1954a,40 D'Amanda 2019a,41 Dreyzin 2014,43 Eiser 2005,44 Elemraid 2013,45 Gill 2014,48 Greenberg 2017,50 Harth 1990,51 Hayman 2001a,53 Helgesson 2009,54 Hoberman 2013a,55 Hoehn 2005,56 Howard Sharp 2020,58 Hulst 2005,59 Ingersgaard 2018,60 Jollye 2009,63 Korotchikova 2010,65 Kumari 2019,66 Langley 1998,68 Mason 2000,69 Menon 2012,71 Menon 2014,72 Mwangi 2017a,75 Nieminen 2015,76 Norris 2010,77 Papaz 2012,79 Paquette 2019,80 Read 2009,81 Shah 2018a,23 Shilling 2009,84 Snowdon 2006,86 Surun 2018,88 Tait 1998,91 Tait 2003a,89 Tait 2004a,90 Thomas 2013,93 Volkening 2017,95 Ward 2009,96 Woodgate 2010,97 Woolfall 2013,98 Zupancic 1997a.24 |
Negative impact on medical care | 21 | Barakat 2014,33 Baxter 2012,35 Braga 2014,36 Buck 2015,37 Clausen 1954,40 D'Amanda 2019,41 Elemraid 2013,45 Gattuso 2006,46 Genetti 2019,47 Gonzalez 2016,49 Helgesson 2009,54 Hoberman 2013a,55 Ingersgaard 2018,60 Menon 2014,72 Mihrshahi 2002,73 Morley 2005,74 Sureshkumar 2012,87 Tait 1998,91 Tait 2003a,89 Taylor 2015,92 Woolfall 2013.98 | |
Lack of direct benefit to child | 16 | Barakat 2014,33 Bartlett 2018,34 Clausen 1954,40 D'Amanda 2019a,41 Gattuso 2006,46 Genetti 2011,47 Gill 2014,48 Greenberg 2017,50 Helgesson 2009,54 Howard Sharp 2020,58 Kick 2018,64 Menon 2014,72 Mihrshahi 2002,73 Read 2009,81 Skinner 2011,85 Sureshkumar 2012.87 | |
Child's own medical concerns | 9 | Chappuy 2006,39 Hulst 2005,59 Menon 2014,72 Mihrshahi 2002,73 Morley 2005,74 Sureshkumar 2012,87 Taylor 2015,92 Thomas 2013,93 Woodgate 2010.97 | |
Phlebotomy concerns | 14 | Chantler 2007,38 D'Amanda 2019a,41 Dlugos 2005,42 Helgesson 2009,54 Jay 2007a,61 Jenkins 2009,62 Kick 2018,64 Labib 2018,67 Mazzocco 1999,70 Menon 2012,71 Menon 2014,72 Scollon 2014,83 Vecchi 2013,94 Wynn 2010.99 | |
Burdens | Logistics or burden of participation | 35 | Bartlett 2018,34 Baxter 2012,35 Chappuy 2006,39 D'Amanda 2019a,41 Gattuso 2006,46 Genetti 2019,47 Gill 2014,48 Gonzalez 2016,49 Greenberg 2017,50 Harth 1990,51 Hayman 2001,53 Helgesson 2009,54 Hoehn 2009,56 Hulst 2005,59 Jenkins 2009,62 Kick 2018,64 Korotchikova 2010,65 Langley 1998,68 Mason 2000,69 Mazzocco 1999,70 Mihrshahi 2002,73 Papaz 2012,79 Paquette 2019,80 Read 2009,81 Shilling 2009,84 Skinner 2011,85 Sureshkumar 2012,87 Tait 1998,91 Tait 2003a,89 Taylor 2015,92 Vecchi 2013,94 Volkening 2017,95 Woolfall 2013,98 Wynn 2010,99 Zupancic 1997a.24 |
Lack of trust | Lack of trust in clinicians | 1 | Chappuy 2006.39 |
Lack of trust in medical researchers | 6 | Greenberg 2017,50 Helgesson 2009,54 Mason 2000,69 Read 2009,81 Shah 2018a,23 Wynn 2010.99 | |
Lack of trust in particular institution | 2 | Harth 1990,51 Morley 2005.74 | |
Lack of trust in medicine in general | 2 | Buck 2015,37 Harth 1990.51 | |
Lack of trust in research | 4 | Chantler 2007,38 Dreyzin 2014,43 Mazzocco 1999,70 Snowdon 2006.86 | |
General research concerns | Lack of interest in research | 18 | Braga 2014,36 Elemraid 2013,45 Gattuso 2006,46 Genetti 2019,47 Gonzalez 2016,49 Harth 1990,51 Hulst 2005,59 Langley 1998,68 Mazzocco 1999,70 Menon 2012,71 Menon 2014,72 Mihrshahi 2002,73 Morley 2005,74 Papaz 2012,79 Sammons 2007,82 Skinner 2011,85 Taylor 2015,92 Volkening 2017.95 |
Guinea pig concerns | 8 | Buck 2015,37 Chantler 2007,38 Clausen 1954,40 Greenberg 2017,50 Hoehn 2005,56 Korotchikova 2010,65 Shah 2018a,23 Tait 1998.91 | |
Privacy concerns | 12 | Gattuso 2006,46 Genetti 2019,47 Gill 2014,48 Harth 1990,51 Howard Sharp 2020,58 Kick 2018,64 Kumari 2019,66 Menon 2012,71 Papaz 2012,79 Sammons 2007,82 Scollon 2014,83 Tait 2004.90 | |
Lack of belief in research | 6 | Helgesson 2009,44 Hulst 2005,59 Labib 2018,67 Nieminen 2015,76 Taylor 2015,92 Tait 1998a.91 | |
Discomfort with randomization | 18 | Braga 2014,54 Buck 2015,37 Eiser 2005,76 Gonzalez 2016,49 Greenberg 2017,50 Hoberman 2013a,55 Ingersgaard 2018,60 Jollye 2009,63 Kick 2018,64 Nieminen 2015,76 Norris 2010,77 Sammons 2007,82 Snowdon 2006,86 Surun 2018,88 Tait 1998,91 Woodgate 2010,97 Woolfall 2013,98 Wynn 2010.99 | |
Genetic testing concerns | 5 | Genetti 2019,47 Howard Sharp 2020,58 Papaz 2012,79 Scollon 2014,83 Skinner 2011.85 | |
Informational and consent process related | Not enough information provided about study; inadequate understanding of study | 16 | Barakat 2014,33 Chappuy 2006,39 Dahan 2020,22 Dreyzin 2014,43 Eiser 2005,44 Gill 2014,48 Helgesson 2009,54 Hoberman 2013a,55 Korotchikova 2010,65 Menon 2012,71 Menon 2014,72 Mwangi 2017a,75 Paquette 2019,80 Read 2009,81 Tait 2003a,89 Tait 2004.90 |
Not enough time to make decision about enrollment | 13 | Barakat 2014,33 Chappuy 2006,39 Eiser 2005,44 Hoehn 2009a,56 Jollye 2009,63 Menon 2012,71 Papaz 2012,79 Paquette 2019,80 Snowdon 2006,86 Tait 1998,91 Tait 2004,90 Thomas 2013,93 Ward 2009.96 | |
Not enough knowledge about medical condition or lack of comfort with clinical decision | 2 | Mazzocco 1999,70 Read 2009a.81 | |
Decision too stressful | 29 | Dahan 2020,22 Dlugos 2005,42 Genetti 2019,47 Greenberg 2017,50 Hoberman 2013a,55 Howard Sharp 2020,58 Hulst 2005,59 Ingersgaard 2018,60 Jollye 2009,63 Labib 2018,67 Mason 2000,69 Menon 2012,71 Menon 2014,72 Norris 2010,77 Papaz 2012,79 Paquette 2019,80 Read 2009,81 Sammons 2007,82 Scollon 2014,83 Shah 2018a,23 Snowdon 2006,86 Tait 1998a,91 Tait 2003a,90 Tait 2004,89 Thomas 2013,93 Ward 2009,96 Woodgate 2010,97 Woolfall 2013,98 Zupancic 1997a.24 | |
Parental perception of illness | 10 | Barakat 2014,33 Bartlett 2018,34 Buck 2015,37 Chantler 2007a,38 Clausen 1954,40 D'Amanda 2019a,41 Dreyzin 2014a,43 Jollye 2009,63 Wynn 2010,99 Zupancic 1997a.24 | |
Language barrier | 2 | Helgesson 2009,54 Papaz 2012.79 | |
Prior experience or lack of experience with research | 13 | Chantler 2007a,38 Dahan 2020,22 Gonzalez 2016,49 Hulst 2005,59 Menon 2012,71 Menon 2014,72 Nieminen 2015a,76 Paquette 2019a,80 Shah 2018a,23 Tait 1998a,91 Tait 2003a,90 Tait 2004,89 Thomas 2013a.93 | |
Relational | Discomfort with proxy nature of consent | 3 | Chantler 2007,38 Papaz 2012,79 Woodgate 2010.97 |
One parent wanted to enroll, other did not | 8 | Dahan 2020,22 Genetti 2019,47 Hoehn 2009,56 Jenkins 2009,62 Korotchikova 2010a,65 Langley 1998,68 Menon 2014,72 Skinner 2011.85 | |
Family problems | 5 | Baxter 2012,35 Chantler 2007,38 Jay 2007a,61 Menon 2012,71 Mihrshahi 2002.73 | |
Feeling pressured | 2 | Hayman 2001a,53 Mason 2000.69 | |
Child's wishes related to enrollment | 15 | Chantler 2007,38 Chappuy 2006,39 Clausen 1954,40 Gill 2014,48 Greenberg 2017,50 Howard Sharp 2020,58 Hulst 2005,59 Ingersgaard 2018,60 Jay 2007a,61 Menon 2014,72 Shilling 2009,84 Sureshkumar 2012,87 Tait 2003a,90 Woodgate 2010,97 Woolfall 2013.98 | |
Decision-making style | 1 | Hoberman 2013a55 |
Study directly compared by enrollment status. See Table 9 for more details.
Comparison of Barriers for Participation in Pediatric Research Between Participants and Nonparticipants
Category . | Barrier . | Study . | Discussion . | Statistics . |
---|---|---|---|---|
Concerns about study participation | Risk to child | Clausen 1954d,40 | More mothers who gave consent (82%) than mothers who withheld consent (18%) reported being “completely convinced that the shots will be perfectly safe” | NA |
D’Amanda 2019d,41 | More parents who declined enrollment than those who enrolled reported perceiving study as high risk | NA | ||
dMore decliners (4/15) than joiners (1/16) expressed concerns that the experimental drug would be ineffective or have side effects | NA | |||
Hayman 2001d,53 | More declining parents (29%) than consenters (13%) were concerned about safety of test | NA | ||
Hoberman 2013e,55 | Nonconsenters were more likely than consenters to cite the “risk of being in the study” as a barrier to participation | P < .001 | ||
Mwangi 2017d,75 | More decliners (8/15) than participants (1/20) “discussed potential risks to child of participating” | NA | ||
Shah 2018d,23 | Decliners (43/58) were less likely than enrollers (90/105) to rate “the possible risks associated with Epo” as “very important or important” to their decision making | P = .09 | ||
Tait 2003e,89 | Nonconsenters had higher perception of study risk (4.3 on scale of 1–10) than consenters (1.5) | P < .001 | ||
Tait 2004e,90 | Nonconsenters were more likely than consenters to view the risk-benefit ratio as higher | P < .001 | ||
Zupancic 1997e,24 | Nonconsenters assessed study as of higher risk and lower benefit compared with consenters | P < .0001 | ||
Negative impact on medical care | Hoberman 2013e,55 | Nonconsenters were more likely than consenters to cite concern that the “study would interfere with the standard care” | P < .001 | |
Tait 2003e89 | Nonconsenters (47.2%) were more concerned than consenters (8%) that participation would affect child's care | P < .0001 | ||
Lack of direct benefit to child | D’Amanda 2019d,41 | More decliners (11/15) than joiners (2/16) had no expectation of individual benefit | NA | |
Phlebotomy concerns | D’Amanda 2019b,41 | More joiners (9/16) than decliners (4/15) cited blood draw requirements as a barrier | NA | |
Jay 2007e,61 | Nonconsenters were more likely than consenters to worry about their child having a blood test | P = .002 | ||
Burdens | Logistics or burden of participation | D’Amanda 2019d,41 | More decliners (15/15) than joiners (5/16) cited logistical inconvenience as a concern | NA |
Tait 2003e,89 | Nonconsenters (44.6%) were more likely than consenters (8.6%) to view the study as “just another thing to worry about” | P < .0001 | ||
Zupancic 1997d,24 | More nonconsenters (22%) than consenters (10%) agreed or strongly agreed that “the process was too complex” | NA | ||
Lack of trust | Lack of trust in medical researchers | Shah 2018e,23 | Decliners (6/56) were more likely than enrollers (0/103) to report “not trusting” the person who approached them | P = .001 |
General research concerns | Guinea pig concerns | Shah 2018c,23 | Decliners (38/58) did not differ from enrollers (61/105) in rating “the thought of my child being experimented on” as “very important or important” | P = .21 |
Lack of belief in research | Tait 1998e,91 | Nonconsenters were more likely than consenters to have “no opinion” about medical research | P < .01 | |
Discomfort with randomization | Hoberman 2013e,55 | Nonconsenters were more likely than consenters to cite randomization as a barrier to participation | P < .004 | |
eNonconsenters were more likely than consenters to cite parent blinding as a barrier to participation | P < .001 | |||
Informational and consent process related | Not enough information provided about study; inadequate understanding of study | Hoberman 2013c,55 | Consent document characteristics were not different between consenters and nonconsenters | P = .30 |
cConsent environment was not different between consenters and nonconsenters | P = .19 | |||
Mwangi 2017b,75 | More decliners (11/15) than participants (2/20) “demonstrated an understanding of the trial as experimental” | NA | ||
Tait 2003e,89 | Non-consenters (59%) were less likely than consenters (89%) to believe they had been given “just the right amount of information” | P < .0001 | ||
eeeeee | Nonconsenters (34.9%) were more likely than consenters (8.8%) to believe they had been given “too little” information | P < .0001 | ||
Nonconsenters (44.2%) were less likely than consenters (63%) to think the information was “very clear” | P < .0001 | |||
Nonconsenters had lower scores on understanding of the study (19.4 on scale 5–25) compared with consenters (21.4) | P < .001 | |||
Nonconsenters (59%) were less likely than consenters (89%) to believe that they had been given “just the right amount of information” | P < .0001 | |||
Nonconsenters (34.9%) were less likely than consenters (8.8%) to believe they had been given “too little” information | P < .0001 | |||
Nonconsenters (44.2%) were less likely than consenters (63%) to think that the information was “very clear” | P < .0001 | |||
Not enough time to make decision about enrollment | Hoehn 2009d,57 | Fewer nonconsenters than consenter perceived adequacy of time to make decision | NA | |
Not enough knowledge about medical condition or lack of comfort with clinical decision | Read 2009c,81 | Enrollment rate did not differ based on “comfort with the medical language.” | P = NS | |
Decision too stressful | Hoberman 2013e,55 | Nonconsenters reported more anxiety about decision and difficulty making the decision than consenters | P < .001 | |
eNonconsenters had higher “uncertainty” scores than consenters | P < .001 | |||
Shah 2018e,23 | Decliners (21.9 on scale 0–100) had higher decisional conflict scores than enrollers (17.2) | P = .03 | ||
Tait 2003e,89 | Nonconsenters (31.2%) were more likely than consenters (8.8%) to report being anxious as a result of being asked to have their child participate | P < .0001 | ||
e | Nonconsenters had greater decisional uncertainty (7.2 on scale 3–15) than consenters (6.2) | P < .001 | ||
c | Nonconsenters had similar anxiety score (7.3 on scale 1–10) compared with consenters (6.5) | P = NS | ||
Tait 1998e,91 | Nonconsenter (21.1%) were less likely than consenters (6%) to report that the researcher seeking consent had made them feel more anxious | P < .01 | ||
Zupancic 1997d,24 | More nonconsenters (6%) than consenters (3%) reported feeling pressure to consent | NA | ||
Parental perception of illness | Chantler 2007d,38 | More parents who enrolled than who declined enrollment reported perceiving their child as healthy and able to handle the study procedures | NA | |
D’Amanda 2019d,41 | More decliners (53%) than joiners (19%) cited autism as a comorbidity factor as a barrier | NA | ||
Dreyzin 2014b,43 | More decliners (1/1) than consenters (0/5) “perceived their child's health as relatively stable” | NA | ||
Zupancic 1997c,24 | Parental perceptions of illness severity were similar between consenters and non-consenters | NA | ||
Prior experience with research or the medical system | Chantler 2007c,38 | Variable familiarity with science and medicine was seen in both parents who enrolled and those who declined enrollment | NA | |
Nieminen 2015a,76 | Nonconsenters (282/356) were more likely than consenters (642/965) to report no previous experience with medical trials | P < .001 | ||
Paquette 2019b,80 | There was increased enrollment among those with prior research participation | P = .08 | ||
Shah 2018c,23 | Decliners did not differ from enrollers in prior research participation | P = NS | ||
Tait 2003c,89 | Nonconsenters (18.3%) did not differ from consenters (20.0%) in prior research experience of the child | P = NS | ||
c | Nonconsenters (21.5%) did not differ from consenters (24.4%) in prior research experience of the parent | P = NS | ||
Thomas 2013c,93 | Past experience with research was seen in both parents who enrolled and those who declined enrollment | NA | ||
Tait 1998a,91 | Nonconsenters were less likely to have had their child participate in a previous study | P < .05 | ||
Relational | One parent wanted to enroll, other did not | Korotchikova 2010e,65 | Parents were more likely to consent if both parents were present (96%) compared with when only mother was present (53%) | P < .0001 |
Family problems | Jay 2007e,61 | Nonconsenters were more likely than consenters to report a “presence of other personal reasons” | P = .0001 | |
Feeling pressured | Hayman 2001b,53 | Fewer decliners (2%) than consenters (5%) reported feeling pressured into taking part in study | NA | |
Child's wishes related to enrollment | Jay 2007d,61 | More nonconsenters than consenters to “reported that discussing the follow-up [requirements] with their child influenced their decision to participate” | NA | |
eTait 2003c,89 | Nonconsenters (67%) were no more likely than consenters (48%) to state that their child influenced their decision | P = NS | ||
Decision -making style | Hoberman 2013c,55 | Decision making style (family or self versus shared with doctor) did not differ between consenters and non-consenters | P = NS |
Category . | Barrier . | Study . | Discussion . | Statistics . |
---|---|---|---|---|
Concerns about study participation | Risk to child | Clausen 1954d,40 | More mothers who gave consent (82%) than mothers who withheld consent (18%) reported being “completely convinced that the shots will be perfectly safe” | NA |
D’Amanda 2019d,41 | More parents who declined enrollment than those who enrolled reported perceiving study as high risk | NA | ||
dMore decliners (4/15) than joiners (1/16) expressed concerns that the experimental drug would be ineffective or have side effects | NA | |||
Hayman 2001d,53 | More declining parents (29%) than consenters (13%) were concerned about safety of test | NA | ||
Hoberman 2013e,55 | Nonconsenters were more likely than consenters to cite the “risk of being in the study” as a barrier to participation | P < .001 | ||
Mwangi 2017d,75 | More decliners (8/15) than participants (1/20) “discussed potential risks to child of participating” | NA | ||
Shah 2018d,23 | Decliners (43/58) were less likely than enrollers (90/105) to rate “the possible risks associated with Epo” as “very important or important” to their decision making | P = .09 | ||
Tait 2003e,89 | Nonconsenters had higher perception of study risk (4.3 on scale of 1–10) than consenters (1.5) | P < .001 | ||
Tait 2004e,90 | Nonconsenters were more likely than consenters to view the risk-benefit ratio as higher | P < .001 | ||
Zupancic 1997e,24 | Nonconsenters assessed study as of higher risk and lower benefit compared with consenters | P < .0001 | ||
Negative impact on medical care | Hoberman 2013e,55 | Nonconsenters were more likely than consenters to cite concern that the “study would interfere with the standard care” | P < .001 | |
Tait 2003e89 | Nonconsenters (47.2%) were more concerned than consenters (8%) that participation would affect child's care | P < .0001 | ||
Lack of direct benefit to child | D’Amanda 2019d,41 | More decliners (11/15) than joiners (2/16) had no expectation of individual benefit | NA | |
Phlebotomy concerns | D’Amanda 2019b,41 | More joiners (9/16) than decliners (4/15) cited blood draw requirements as a barrier | NA | |
Jay 2007e,61 | Nonconsenters were more likely than consenters to worry about their child having a blood test | P = .002 | ||
Burdens | Logistics or burden of participation | D’Amanda 2019d,41 | More decliners (15/15) than joiners (5/16) cited logistical inconvenience as a concern | NA |
Tait 2003e,89 | Nonconsenters (44.6%) were more likely than consenters (8.6%) to view the study as “just another thing to worry about” | P < .0001 | ||
Zupancic 1997d,24 | More nonconsenters (22%) than consenters (10%) agreed or strongly agreed that “the process was too complex” | NA | ||
Lack of trust | Lack of trust in medical researchers | Shah 2018e,23 | Decliners (6/56) were more likely than enrollers (0/103) to report “not trusting” the person who approached them | P = .001 |
General research concerns | Guinea pig concerns | Shah 2018c,23 | Decliners (38/58) did not differ from enrollers (61/105) in rating “the thought of my child being experimented on” as “very important or important” | P = .21 |
Lack of belief in research | Tait 1998e,91 | Nonconsenters were more likely than consenters to have “no opinion” about medical research | P < .01 | |
Discomfort with randomization | Hoberman 2013e,55 | Nonconsenters were more likely than consenters to cite randomization as a barrier to participation | P < .004 | |
eNonconsenters were more likely than consenters to cite parent blinding as a barrier to participation | P < .001 | |||
Informational and consent process related | Not enough information provided about study; inadequate understanding of study | Hoberman 2013c,55 | Consent document characteristics were not different between consenters and nonconsenters | P = .30 |
cConsent environment was not different between consenters and nonconsenters | P = .19 | |||
Mwangi 2017b,75 | More decliners (11/15) than participants (2/20) “demonstrated an understanding of the trial as experimental” | NA | ||
Tait 2003e,89 | Non-consenters (59%) were less likely than consenters (89%) to believe they had been given “just the right amount of information” | P < .0001 | ||
eeeeee | Nonconsenters (34.9%) were more likely than consenters (8.8%) to believe they had been given “too little” information | P < .0001 | ||
Nonconsenters (44.2%) were less likely than consenters (63%) to think the information was “very clear” | P < .0001 | |||
Nonconsenters had lower scores on understanding of the study (19.4 on scale 5–25) compared with consenters (21.4) | P < .001 | |||
Nonconsenters (59%) were less likely than consenters (89%) to believe that they had been given “just the right amount of information” | P < .0001 | |||
Nonconsenters (34.9%) were less likely than consenters (8.8%) to believe they had been given “too little” information | P < .0001 | |||
Nonconsenters (44.2%) were less likely than consenters (63%) to think that the information was “very clear” | P < .0001 | |||
Not enough time to make decision about enrollment | Hoehn 2009d,57 | Fewer nonconsenters than consenter perceived adequacy of time to make decision | NA | |
Not enough knowledge about medical condition or lack of comfort with clinical decision | Read 2009c,81 | Enrollment rate did not differ based on “comfort with the medical language.” | P = NS | |
Decision too stressful | Hoberman 2013e,55 | Nonconsenters reported more anxiety about decision and difficulty making the decision than consenters | P < .001 | |
eNonconsenters had higher “uncertainty” scores than consenters | P < .001 | |||
Shah 2018e,23 | Decliners (21.9 on scale 0–100) had higher decisional conflict scores than enrollers (17.2) | P = .03 | ||
Tait 2003e,89 | Nonconsenters (31.2%) were more likely than consenters (8.8%) to report being anxious as a result of being asked to have their child participate | P < .0001 | ||
e | Nonconsenters had greater decisional uncertainty (7.2 on scale 3–15) than consenters (6.2) | P < .001 | ||
c | Nonconsenters had similar anxiety score (7.3 on scale 1–10) compared with consenters (6.5) | P = NS | ||
Tait 1998e,91 | Nonconsenter (21.1%) were less likely than consenters (6%) to report that the researcher seeking consent had made them feel more anxious | P < .01 | ||
Zupancic 1997d,24 | More nonconsenters (6%) than consenters (3%) reported feeling pressure to consent | NA | ||
Parental perception of illness | Chantler 2007d,38 | More parents who enrolled than who declined enrollment reported perceiving their child as healthy and able to handle the study procedures | NA | |
D’Amanda 2019d,41 | More decliners (53%) than joiners (19%) cited autism as a comorbidity factor as a barrier | NA | ||
Dreyzin 2014b,43 | More decliners (1/1) than consenters (0/5) “perceived their child's health as relatively stable” | NA | ||
Zupancic 1997c,24 | Parental perceptions of illness severity were similar between consenters and non-consenters | NA | ||
Prior experience with research or the medical system | Chantler 2007c,38 | Variable familiarity with science and medicine was seen in both parents who enrolled and those who declined enrollment | NA | |
Nieminen 2015a,76 | Nonconsenters (282/356) were more likely than consenters (642/965) to report no previous experience with medical trials | P < .001 | ||
Paquette 2019b,80 | There was increased enrollment among those with prior research participation | P = .08 | ||
Shah 2018c,23 | Decliners did not differ from enrollers in prior research participation | P = NS | ||
Tait 2003c,89 | Nonconsenters (18.3%) did not differ from consenters (20.0%) in prior research experience of the child | P = NS | ||
c | Nonconsenters (21.5%) did not differ from consenters (24.4%) in prior research experience of the parent | P = NS | ||
Thomas 2013c,93 | Past experience with research was seen in both parents who enrolled and those who declined enrollment | NA | ||
Tait 1998a,91 | Nonconsenters were less likely to have had their child participate in a previous study | P < .05 | ||
Relational | One parent wanted to enroll, other did not | Korotchikova 2010e,65 | Parents were more likely to consent if both parents were present (96%) compared with when only mother was present (53%) | P < .0001 |
Family problems | Jay 2007e,61 | Nonconsenters were more likely than consenters to report a “presence of other personal reasons” | P = .0001 | |
Feeling pressured | Hayman 2001b,53 | Fewer decliners (2%) than consenters (5%) reported feeling pressured into taking part in study | NA | |
Child's wishes related to enrollment | Jay 2007d,61 | More nonconsenters than consenters to “reported that discussing the follow-up [requirements] with their child influenced their decision to participate” | NA | |
eTait 2003c,89 | Nonconsenters (67%) were no more likely than consenters (48%) to state that their child influenced their decision | P = NS | ||
Decision -making style | Hoberman 2013c,55 | Decision making style (family or self versus shared with doctor) did not differ between consenters and non-consenters | P = NS |
NA, not applicable; NS, not significant.
Significantly associated with enrolling (quantitative only: P < .05)
Tends toward enrolling (quantitative: [P ≥ .05 and P ≤ .1] or qualitative)
No difference (quantitative or qualitative)
Tends toward declining (quantitative: (P ≥ .05 and P ≤ .1) or qualitative)
Significantly associated with declining (quantitative only: P < .05)
Concerns About Study Participation
Concerns about study participation emerged as an important category of barriers to enrollment. Here we included only direct negative impacts of research participation. These included risk to child, negative impact on patient care, lack of direct benefit to child, the child’s own medical concerns, and phlebotomy concerns. The most frequently mentioned barriers to study participation in this category were risk to child (N = 46), negative impact on medical care (N = 21) and lack of direct benefit to child (N = 16). In each of these, all studies comparing by enrollment status identified a negative association between the barrier and enrollment. Nine studies reported the child’s own medical concerns (eg, the child does not know their true diagnosis or prognosis) as a barrier to enrollment, though none compared by enrollment status. Fourteen studies reported phlebotomy concerns as a barrier to enrollment; this was variably associated with enrollment.
Burdens
Burdens of participation were identified as barriers to pediatric research participation. Within this category, we included the logistical burden of participation. Studies frequently reported the logistical burden of participation (N = 35) as a barrier to enrollment; this was the second most reported barrier in any category surpassed only by risk to child. All 3 studies that reported the logistical burden of participation as a barrier found a negative association with this barrier and enrollment.
Lack of Trust
Lack of trust, distrust, and mistrust were important barriers to enrollment. Because of the heterogeneity of how these terms were defined and used, we did not distinguish between them. We did differentiate between the object of low trust: lack of trust in medical researchers (N = 6), lack of trust in medical research (N = 4), lack of trust in a particular institution (N = 2), lack of trust in medicine in general (N = 2), and lack of trust in clinicians (N = 1). The single study that compared by enrollment status found that lower trust in medical research was associated with lower enrollment.
General Research Concerns
Another important theme of barriers to enrollment was general concern with research. Within this category, we included: lack of interest in research, concern over their child being a “guinea pig,” privacy concerns, lack of belief in research, discomfort with randomization, and genetic testing concerns. Whereas there is some overlap between lack of interest in research and lack of belief in research, there was enough distinction to create separate barriers. Lack of interest captured a current lack of interest in research, whereas lack of belief reflected a moral objection or belief that research does not work in general. Definitions and examples are provided in Table 5. Studies frequently reported lack of interest in research (N = 18), discomfort with randomization (N = 18) and lack of belief in research (N = 6) as barriers; all studies that compared by enrollment status found a negative association with the barrier. Twelve studies reported privacy concerns, but none compared by enrollment status.
Informational and Consent Process Related
Concerns related to the consent process or information sharing emerged as important barriers to enrollment. This large category included 7 items (Table 5). Studies frequently reported these barriers to enrollment: inadequate understanding of study (N = 16), not enough time to make decision (N = 13), and decision being too stressful (N = 29). For each of these, most studies that compared by enrollment status found a negative association between the barrier and enrollment. Ten studies found that parent perception of illness was a barrier to enrollment. In the 4 studies that compared by enrollment status, the barrier was variably associated with enrollment: 2 studies found that lower parental perception of illness was associated with lower enrollment, 1 study found that lower parent perception of illness was associated with higher enrollment, and 1 study found no association.
Relational
Concerns related to parental relationships emerged as barriers to enrollment. These included 6 items (Table 5). Most commonly reported were child’s wishes to not participate (N = 15) and differing preferences between parents (N = 8). Few studies compared these items directly by enrollment status.
Changes Over Time
Barriers to research were examined across decades to see if any changes emerged. The most cited barriers to enrollment in each of these 3 epochs (1990s, 2000s, 2010s) were logistical burdens (N = 5, N = 11, N = 19), risk to child (N = 4, N = 14, N = 26), and the decision being too stressful (N = 2, N = 10, N = 15). These 3 categories were similarly cited over time, however 2 less cited categories had more citations in the most recent cohort: discomfort with randomization (N = 1, N = 4, N = 13) and concerns related to negative impact on medical care (N = 1, N = 5, N = 14).
Enrollment Rate and Population Size
Parental inclusion rates within the enrollment studies included in this review is challenging to interpret because of variable reporting. Several studies (N = 7) did not report the number of included parents. The studies that did report included parents often did not report number of parents eligible but not included in the enrollment study.
However, 2 notable trends emerged from the available data (Table 10). First, inclusion rates varied drastically by type of enrollment study: surveys and interviews included less than half of eligible parents, whereas recruitment log studies included more than 90%. Second, inclusion rates varied by enrollment status, with greater inclusion among parents who enrolled in the feeder study compared with those who declined participation in the feeder study.
Overall Enrollment Rate and Population Size of Enrollment Studies
Enrollment Study Type . | Manuscripts . | Enrolled in Feeder Study . | Declined Enrollment in Feeder Study . | ||
---|---|---|---|---|---|
N Included in Enrollment Study . | N Included or Eligible (%) for Enrollment Study . | N Included in Enrollment Study . | N Included or Eligible (%) for Enrollment Study . | ||
Survey and interview studies | 46a | 5800 | 3044/6174 (49%)b | 2239 | 1394/3846 (36%)b |
Recruitment log studies | 24 | 10 218 | 10 218/10 551 (97%)c | 15 495 | 15 403/17 175 (90%)c |
All studies | 70 | 16 018 | 13 262/16 725 (79%)d | 17 734 | 16 797/21 021 (80%)d |
Enrollment Study Type . | Manuscripts . | Enrolled in Feeder Study . | Declined Enrollment in Feeder Study . | ||
---|---|---|---|---|---|
N Included in Enrollment Study . | N Included or Eligible (%) for Enrollment Study . | N Included in Enrollment Study . | N Included or Eligible (%) for Enrollment Study . | ||
Survey and interview studies | 46a | 5800 | 3044/6174 (49%)b | 2239 | 1394/3846 (36%)b |
Recruitment log studies | 24 | 10 218 | 10 218/10 551 (97%)c | 15 495 | 15 403/17 175 (90%)c |
All studies | 70 | 16 018 | 13 262/16 725 (79%)d | 17 734 | 16 797/21 021 (80%)d |
Includes 7 studies that did not describe enrollment numbers.
Includes the 16 out of 46 manuscripts that reported N eligible for the enrollment study.
Includes the 23 out of 24 manuscripts that reported N eligible for the enrollment study.
Includes the 39 out of 70 manuscripts that reported N eligible for the enrollment study.
Discussion
In this review of barriers and facilitators to research participation in pediatrics, our findings suggest several conclusions we will discuss. First, health benefit to child and altruism emerged as the most consistent motivators for study participation. Second, leading barriers to enrollment were a potential risk to child and the burdens of participation. Third, parental perception of illness was associated with enrollment in complex ways. Fourth, even with a review methodology that prioritized identifying the views of research decliners, the information available from this elusive population was limited. Lastly, we will consider how societal events in recent years may contribute to changes in the importance of facilitators and barriers to pediatric research participation.
Health benefit to child and altruism were the most identified facilitators to research participation. This is consistent with an earlier review, which also identified personal health benefit for child and altruism as the most cited motivating factors for enrollment.26 The relationship between perceived health benefit to child and research participation is an ethically fraught and complicated one. If parental perception of health benefit to child results in increased enrollment, then researchers should promote the health benefits of research participation. However, this may be problematic if the perception of benefit is incorrect. Sometimes, participation in research may result in health benefits to the child, such as access to an intervention otherwise unavailable.100 More often, however, there may not be a direct benefit to the child, or the potential for direct benefit may be uncertain.14 Our methods are not able to determine how much such a belief in potential benefit to your child may overlap with the therapeutic misconception in which research participants believe that the goal of research is to benefit participants, rather than to generate generalizable knowledge.101 This may be particularly problematic in high-stakes scenarios.102 Future work must access ways to harness this facilitator of research participation without misleading parents considering research for their child.
Altruism emerged as another strong facilitator to research participation. There is a growing body of literature suggesting that multiple kinds of altruism may exist. Research altruism may be a specific kind of altruism different from other types.103 Others have proposed a phenomenon of “conditional altruism,” which may prime interest in research participation but is not sufficient to result in participation without other factors.104 The included manuscripts do not shed light on whether research participation related altruism may be a modifiable factor. Future work should identify how altruism may be harnessed to optimize participation within pediatric research.
The potential risk to child emerged as the most mentioned barrier to participation. Tromp similarly found that fear of risks is the most frequent discouraging factor to participation in clinical trials.26 Medical risks of study participation was the most cited reason for those declining an adult statin clinical trial105 and a pediatric sickle cell clinical trial.106 This suggests the need for improvements in how researchers inform parents of potential risks to child and in strategies to effectively address parental concerns.
Burden of participation was the second most frequently cited barrier, followed closely by concerns that the decision was too stressful. This is consistent with a review of qualitative studies asking adult research participants (excluding decliners) about their experiences that found that the psychological burdens of participation were substantial and were present through all parts of the research process.107 A review of qualitative studies of adult research participants and decliners identified wariness of additional burden of research participation as a major theme.108 Researchers may find it beneficial to consider methods to mitigate burdens of participation, both psychological and logistical, to improve recruitment in clinical trials. These may include assistance or reimbursement for travel, childcare, use of virtual research visits or home visits, coordination of study visits and laboratory draws with other clinic appointments, and increased availability of translation services. Such efforts have the potential to ease the burden of a clinical trial with the potential to increase participation. Importantly, these burdens are most impactful for under-represented populations or for those with limited access to resources. Addressing burdens must be approached with the goal of decreasing inequities in research participation.
Parental perception of illness emerged as a barrier that was associated with enrollment status in complex ways. Illness perception is a complex phenomenon, impacted not only by clinical situation but by interpretation of that situation by a parent, which is likely impacted by many things, including health status, familiarity with the disease, and personal factors, such as coping mechanisms. In a study assessing participation in research related to pediatric diabetic ketoacidosis, severity was not associated with enrollment status, but parents of children with previous episodes of ketoacidosis were more likely to enroll in the study than those without past events.109 We found that perception that a child is very ill might serve as either a facilitator or a barrier to enrollment. Fragile or worsening health might be a barrier when a parent perceives the child is not in adequate health to assume the risks of the study. Alternatively, parental perception that a child is in poor or deteriorating health might facilitate trial participation when a parent believes the study may provide a cure or successful treatment of their sick child.110 Further studies regarding factors that determine parental perception of illness and how risk and benefit are viewed, in light of this perception, could increase trial participation.
Although our methods aimed to prioritize research decliners, the data we identified from this population represented well under half of the included population, which limited our ability to directly compare parents who declined pediatric research with those who enrolled their child. This was true for several reasons. First, many studies included far greater numbers of parents who enrolled their child compared with parents who declined participation. Second, many studies that did include higher numbers of decliners reported less useful data from them (eg, screening log reports only) that did not allow for robust analysis of potential facilitators and barriers. Lastly, the methods of many of the included studies did not allow for direct comparison between enrolled and declined parents. For example, some studies only asked those who enrolled about facilitators and only asked those who declined about barriers. Future studies that ask precisely the same questions to parents regardless of enrollment status will enable better understanding of the reasons parents choose to enroll or decline participation in pediatric research.
Lastly, we would like to acknowledge that recent significant societal events may be influencing facilitators and barriers to pediatric research participation. These include but are not limited to: the murder of George Floyd and the Black Lives Matter movement,111 the coronavirus disease 2019 pandemic,112 increasing political polarization,113 global wars,114 and the increasing impact of climate change.115,116 The results from this review revealed no clear trends in the most cited barriers and facilitators in the 1990s, 2000s, and 2010s. However, there is a delay between change in attitudes and the report of such changes in the scientific literature, which precludes the current manuscript from evaluating the potential impact of recent societal events. It is possible, for example, that coronavirus disease 2019 vaccine and mask mandates may result in decreased trust of researchers and clinicians. Many of these events may erode public trust in the police, legal, and political systems, which may diminish trust in medical institutions. Altruistic motivation may be altered by local and global political turmoil. Future work should evaluate how these facilitators and barriers to pediatric research participation continue to change over time.
Limitations
One limitation of our systematic review was that we only included studies in English. Additionally, because of the methods used by the included studies, we were unable to analyze the interactions between factors (eg, how altruism and benefits may together lead toward enrollment decisions) or to rank the relative importance of barriers of facilitators to research participation.
Conclusions
This systematic review offers a comprehensive overview of the barriers and facilitators to participation in pediatric research as reported in peer-reviewed articles. Health benefit to child and altruism emerged as the most cited facilitators. In addition, this review highlighted differences between parents who enroll their child in clinical research and parents who decline to do so.
The findings from this review may guide researchers aiming to create interventions to improve the parental experience of recruitment for pediatric studies and to optimize enrollment rates.
Future work should focus on parents who decline participation in pediatric research and should use methodologies that enable direct comparison between enrolled and declined parents. Additionally, further research is needed to analyze the interactions between barriers and facilitators to research participation, to determine the relative importance of each, and to evaluate how they may interact with other critical variables, such as parent demographics.
Study data were collected and managed using Research Electronic Data Capture (REDCap) electronic data capture tools hosted at the University of Washington.117,118 REDCap is a secure, web-based software platform designed to support data capture for research studies, providing (1) an intuitive interface for validated data capture; (2) audit trails for tracking data manipulation and export procedures; (3) automated export procedures for seamless data downloads to common statistical packages; and (4) procedures for data integration and interoperability with external sources. REDCap at the University of Washington is supported by Institute of Translational Health Science grant support (UL1 TR002319, KL2 TR002317, and TL1 TR002318 from NCATS/NIH).
Acknowledgments
We thank research librarian Peggy Cruse, MA, Seattle Children’s Hospital, for assistance performing our systematic review; Amanda Mercer, BA, Treuman Katz Center for Pediatric Bioethics, for literature review assistance; Sheila Ganti, PhD, Seattle Children’s Hospital, for assistance with REDCap creation; Megan Gray, MD, University of Washington, for assistance with data presentation; and Stephanie Kraft, JD, University of Washington, for critically reviewing a draft of the manuscript.
Dr Weiss conceptualized the study and designed the study; Dr Nathe designed the data collection instruments, collected data, conducted the analysis, and drafted the initial manuscript; Ms Oskoui designed the data collection instruments, collected data, and conducted the analysis; and all authors reviewed and revised the manuscript, approved the final manuscript as submitted, and agree to be accountable for all aspects of the work.
FUNDING: This study was supported in part by the National Institutes of Health through the Eunice Kennedy Shriver National Institute of Child Health and Human Development (K23HD103872). The funders had no control over design and conduct of the study; collection, management, analysis, and interpretation of the data; preparation, review, or approval of the manuscript; and decision to submit the manuscript for publication. The National Institutes of Health had no role in the design and conduct of this study.
CONFLICT OF INTEREST DISCLOSURES: The authors have indicated they have no conflicts of interest relevant to this article to disclose.
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