INTRODUCTION
METHODS
Data sources and search strategy
Inclusion and exclusion criteria
Study selection
Data extraction and study quality assessment
Level of evidence a
| Characteristics |
---|---|
A1 | Systematic reviews involving at least two studies at A2 level, of which the results of separate studies are consistent |
A2 | Randomized comparative clinical studies of good quality (randomized, double-blind controlled trails) of sufficient size and consistency |
B | Randomized clinical trials of mediocre quality, of insufficient size, or other comparative studies |
C | Non-comparative studies |
D | Expert opinion |
Level of evidence a
| Characteristics |
---|---|
++ | Credible meta-synthesis of qualitative studies |
+ | Credible study |
+/- | Study of which credibility is dubious |
- | Study of which credibility is minimal |
Data-synthesis
RESULTS
Study selection
Characteristics of included studies for systematic review
Characteristic | No. of studies | Studies | |
---|---|---|---|
Type of study |
Quantitative study
|
26
|
See categories below
|
- Written questionnaires | 15 | Barakat, 2013; Berg, 2010; Buscariollo, 2012; Cain, 2005; Cherill, 2010; Hoberman, 2013; Read, 2009; Sammons, 2007; Tait, 1998; Tait, 2003; Truong, 2011; Van Stuijvenberg, 1998; Vanhelst, 2013; Wagner, 2006; Zupancic, 1997 | |
- Verbally administered questionnaires | 7 | Baren, 1999; Brody, 2005; Brody, 2012; Harth, 1999; Miller, 2013; Rothmier, 2003; Wendler, 2012 | |
- Secondary analysis of data | 4 | Menon, 2012; Norris, 2010; Peden, 2000; Wynn, 2010 | |
Qualitative study
|
16
|
See categories below
| |
- Interviews | 10 | Barrera, 2005; Broome, 2003; Cartwright, 2011; Koelch, 2009; Liaschenko, 2001; MacNeill, 2013; Masiye, 2008; Patterson, 2014; Pletsch, 2001; Pletsch, 2001 (2); Woodgate, 2010 | |
- Focus groups a
| 3 | Caldwell, 2003; Lebensburger, 2013 | |
- Secondary analysis of data b
| 3 | Deatrick, 2002; Hoehn, 2005; Oppenheim, 2005 | |
Study population | Only parents/caregivers | 26 | Baren, 1999; Buscariollo, 2012; Cartwright, 2011; Caldwell, 2003; Deatrick, 2002; Harth, 1999; Hoehn, 2005; Lebensburger, 2013; Liaschenko, 2001; MacNeill, 2013; Masiye, 2008; Menon, 2012; Oppenheim, 2005; Pletsch, 2001 (2); Pletsch, 2001; Rothmier, 2003; Sammons, 2007; Tait, 1998; Truong, 2011; Tait, 2003; Van Stuijvenberg, 1998; Vanhelst, 2013; Woodgate, 2010; Zupancic, 1997; Wynn, 2010; Hoberman, 2013 |
Only children | 5 | Broome, 2003; Cain, 2005; Cherill, 2010; Koelch, 2009; Miller, 2013 | |
Both | 11 | Barakat, 2013; Barrera, 2005; Berg, 2010; Brody, 2005; Brody, 2012; Norris, 2010; Patterson, 2014; Peden, 2000; Read, 2009; Wagner, 2006; Wendler, 2012 | |
Setting | Oncology | 11 | Barrera, 2005; Berg, 2010; Broome, 2003; Deatrick, 2002; Liaschenko, 2001; Miller, 2013; Oppenheim, 2005; Pletsch, 2001; Read, 2009; Truong, 2011; Woodgate, 2010 |
Diabetes mellitus | 5 | Broome, 2003; Buscariollo, 2012; Cain, 2005; Pletsch, 2001 (2); Pletsch, 2001 | |
Airway diseases | 7 | Barakat, 2013; Brody, 2005; Brody, 2012; Harth, 1999; MacNeill, 2013; Rothmier, 2003; Sammons, 2007 | |
Sickle cell disease | 4 | Barakat, 2013; Lebensburger, 2013; Patterson, 2014; Wynn, 2010 | |
PICU / NICU | 4 | Cartwright, 2011; Hoehn, 2005; Menon, 2012; Zupancic, 1997 | |
Sick and healthy children (not specified) | 4 | Caldwell, 2003; Cherill, 2010; Vanhelst, 2013; Wendler, 2012 | |
Anesthetics | 3 | Peden, 2000; Tait, 1998; Tait, 2003 | |
Emergency department | 2 | Baren, 1999; Van Stuijvenberg, 1998; | |
Psychopharmacology | 2 | Koelch, 2009; Wagner, 2006 | |
Other c
| 3 | Masiye, 2008; Norris, 2010; Hoberman, 2013; | |
Type of drug research | Real life drug study protocol | 33 | Barrera, 2005; Berg, 2010; Broome, 2003; Cain, 2005; Cartwright, 2011; Deatrick, 2002; MacNeill, 2013; Harth, 1999; Hoberman, 2013; Hoehn, 2005; Koelch, 2009; Liaschenko, 2001; Masiye, 2008; Menon, 2012; Miller, 2013; Norris, 2010; Oppenheim, 2005; Peden, 2000; Pletsch, 2001; Pletsch, 2001 (2); Read, 2009; Rothmier, 2003; Sammons, 2007; Tait, 1998; Tait, 2003; Truong, 2011; Van Stuijvenberg, 1998; Vanhelst, 2013; Wagner, 2006; Wendler, 2012; Woodgate, 2010; Wynn, 2010; Zupancic, 1997 |
Drug research in general | 4 | Sammons, 2007; Tait, 1998; Tait, 2003; Van Stuijvenberg, 1998 | |
Hypothetical drug study protocol | 5 | Baren, 1999; Brody, 2005; Brody, 2012; Lebensburger, 2013; Patterson, 2014 | |
Prospect of direct benefit | Only studies with prospect of direct benefit | 22 | Baren, 1999; Brody, 2012; Cain, 2005; Cartwright, 2011; Harth, 1999; Hoberman, 2013; Hoehn, 2005; Koelch, 2009; MacNeill, 2013; Masiye, 2008; Norris, 2010; Patterson, 2014; Peden, 2000; Pletsch, 2001 (2); Rothmier, 2003; Sammons, 2007; Tait, 1998; Tait, 2003; Van Stuijvenberg, 1998; Wagner, 2006; Wynn, 2010; Zupancic, 1997 |
Only studies with no prospect of direct benefit | 5 | Barrera, 2005; Berg, 2010; Deatrick, 2002; Miller, 2013; Oppenheim, 2005 | |
Both | 7 | Broome, 2003; Liaschenko, 2001; Menon, 2012; Pletsch, 2001; Truong, 2011; Vanhelst, 2013; Wendler, 2012 | |
Not specified | 8 | Barakat, 2013; Brody, 2005; Buscariollo, 2012; Caldwell, 2003; Cherill, 2010; Lebensburger, 2013; Read, 2009; Woodgate, 2010 | |
Consenters or non consenters | Only non consenters | 3 | Peden, 2000; Norris, 2010; Menon, 2012 |
Only consenters | 18 | Broome, 2003; Cain, 2005; Cartwright, 2011; Deatrick, 2002; Liaschenko, 2001; Masiye, 2008; Miller, 2013; Oppenheim, 2005; Pletsch, 2001; Pletsch, 2001 (2); Rothmier, 2003; MacNeill, 2013; Truong, 2011; Van Stuijvenberg, 1998; Vanhelst, 2013; Wagner, 2006; Wendler, 2012; Woodgate, 2010; | |
Both | 21 | Barakat, 2013; Baren, 1999; Barrera, 2005; Berg, 2010; Brody, 2005; Brody, 2012; Buscariollo, 2012; Caldwell, 2003; Cherill, 2010; Harth, 1999; Hoberman, 2013; Hoehn, 2005; Koelch, 2009; Lebensburger, 2013; Read, 2009; Sammons, 2007; Tait, 1998; Tait, 2003; Patterson, 2014; Wynn, 2010; Zupancic, 1997 |
Study quality and risk of bias
Qualitative metasummary of motivating factors
Motivating factor | No. of studies (total = 33) | Individual studies |
---|---|---|
Personal health benefit for child a
| 31 | Barakat, 2013; Baren, 1999; Barrera, 2005; Brody, 2005; Brody, 2012; Buscariollo, 2012; Caldwell, 2003; Cartwright, 2011; Deatrick, 2002; Harth, 1999; Hoberman, 2013; Hoehn, 2005; Lebensburger, 2013; Liaschenko, 2001; MacNeill, 2013; Masiye, 2008; Oppenheim, 2005; Patterson, 2014; Pletsch, 2001; Pletsch, 2001 (2); Read, 2009; Sammons, 2007; Tait, 1998; Tait, 2003; Truong, 2011; Van Stuijvenberg, 1998; Vanhelst, 2013; Wagner, 2006; Woodgate, 2010; Wynn, 2010; Zupancic, 1997 |
Altruism b
| 26 | Baren, 1999; Barrera, 2005; Buscariollo, 2012; Caldwell, 2003; Cartwright, 2011; Deatrick, 2002; Harth, 1999; Hoberman, 2013; Hoehn, 2005; Liaschenko, 2001; MacNeill, 2013; Patterson, 2014; Pletsch, 2001; Pletsch, 2001 (2); Read, 2009; Rothmier, 2003; Sammons, 2007; Tait, 1998; Tait, 2003; Truong, 2011; Van Stuijvenberg, 1998; Vanhelst, 2013; Wendler, 2012; Woodgate, 2010; Wynn, 2010; Zupancic, 1997 |
Trust in safety of research | 12 | Barakat, 2013; Buscariollo, 2012; Cartwright, 2011; Harth, 1999; Hoberman, 2013; Hoehn, 2005; MacNeill, 2013; Patterson, 2014; Tait, 1998; Truong, 2011; Vanhelst, 2013; Zupancic, 1997 |
Relation to researcher | 12 | Buscariollo, 2012; Caldwell, 2003; Harth, 1999; Hoberman, 2013; Masiye, 2008; Read, 2009; Sammons, 2007; Tait, 1998; Tait, 2003; Truong, 2011; Woodgate, 2010; Van Stuijvenberg, 1998 |
More contact with medical team | 8 | Buscariollo, 2012; Caldwell, 2003; Harth, 1999; Lebensburger, 2013; MacNeill, 2013; Masiye, 2008; Wynn, 2010; Woodgate, 2010 |
Benefit for parents themselves | 5 | Harth, 1999; Oppenheim, 2005; Rothmier, 2003; Wagner, 2006; Van Stuijvenberg, 1998; |
Minimal burden for child | 4 | Patterson, 2014; Pletsch, 2001 (2); Read, 2009; Woodgate, 2010 |
Financial reimbursement | 5 | Brody, 2012; Buscariollo, 2012; Harth, 1999; Masiye, 2008; Wagner, 2006 |
Felt as only option c
| 4 | Cartwright, 2011; Deatrick, 2002; Liaschenko, 2001; Oppenheim, 2005 |
Influence of family and friends | 3 | Buscariollo, 2012; Harth, 1999; Read, 2009 |
Motivating factor | No. of studies (total = 10) | Individual studies |
---|---|---|
Personal health benefita
| 8 | Barrera, 2005; Brody, 2005; Brody, 2012; Cain, 2005; Miller, 2013; Patterson, 2014; Read, 2009; Wagner, 2006 |
Altruism | 6 | Cain, 2005; Miller, 2013; Patterson, 2014; Read, 2009; Wagner, 2006; Wendler, 2012 |
Increasing comfort | 4 | Cain, 2005; Koelch, 2009; Miller, 2013; Read, 2009 |
Relation to researcher | 3 | Miller, 2013; Read, 2009; Wagner, 2006 |
Influence of family and friends | 3 | Cain, 2005; Read, 2009; Wagner, 2006 |
Financial reimbursement | 3 | Brody, 2005; Brody, 2012; Wagner, 2006 |
Increasing knowledge | 2 | Cain, 2005; Wagner, 2006 |
Curiosity | 2 | Cain, 2005; Koelch, 2009 |
Felt as only option | 1 | Miller, 2013 |
Qualitative metasummary of discouraging factors
Discouraging factor | No. of studies (total = 24) | Individual studies |
---|---|---|
Fear of risks | 14 | Baren, 1999; Brody, 2005; Brody, 2012; Buscariollo, 2012; Caldwell, 2003; Harth, 1999; Hoehn, 2005; Lebensburger, 2013; MacNeill, 2013; Patterson, 2014; Pletsch, 2001 (2); Read, 2009; Tait, 1998; Tait, 2003; |
Distrust in research (“guinea pig”) | 11 | Baren, 1999; Caldwell, 2003; Harth, 1999; Hoehn, 2005; Lebensburger, 2013; Menon, 2012; Peden, 2000; Read, 2009; Sammons, 2007; Tait, 1998; Wynn, 2010 |
Logistics / disruption of daily life a
| 11 | Baren, 1999; Brody, 2005; Caldwell, 2003; Patterson, 2014; Harth, 1999; Lebensburger, 2013; Peden, 2000; Pletsch, 2001; Read, 2009; Tait, 1998; Wynn, 2010 |
Burden for child | 9 | Barrera, 2005; Brody, 2005; Buscariollo, 2012; Menon, 2012; Oppenheim, 2005; Peden, 2000; Pletsch, 2001 (2); Read, 2009; Woodgate, 2010 |
Decision too stressful | 7 | Hoberman, 2013; Lebensburger, 2013; Menon, 2012; Pletsch, 2001; Read, 2009; Sammons, 2007; Tait, 1998 |
Fear of randomization | 6 | Caldwell, 2003; Lebensburger, 2013; MacNeill, 2013; Sammons, 2007; Tait, 1998; Wynn, 2010 |
No direct benefit for child b
| 5 | Baren, 1999; Barrera, 2005; MacNeill, 2013; Read, 2009; Wynn, 2010 |
Financial constraints | 5 | Baren, 1999; Buscariollo, 2012; Harth, 1999; Tait, 1998; Wynn, 2010 |
Discomfort with proxy consent | 2 | Buscariollo, 2012; Caldwell, 2003 |
Discouraging factor | No. of studies (total = 6) | Individual studies |
---|---|---|
Burden / disruption of daily life | 4 | Brody, 2005; Koelch, 2009; Read, 2009; Patterson, 2014 |
Feeling like a “guinea pig” | 3 | Koelch, 2009; Peden, 2000; Read, 2009 |
Fear of risks | 3 | Brody, 2005; Brody, 2012; Patterson, 2014 |
Decision too stressful | 1 | Read, 2009 |
No understanding | 1 | Read, 2009 |
No direct benefit | 1 | Read, 2009 |
Preference for one arm | 1 | Peden, 2000 |