Top

Published in:

Open Access 01-05-2016 | Review

Motivations of children and their parents to participate in drug research: a systematic review

Authors: Krista Tromp, C. Michel Zwaan, Suzanne van de Vathorst

Published in: European Journal of Pediatrics | Issue 5/2016

ABSTRACT

Information on motivations for research participation, may enable professionals to better tailor the process of recruitment and informed consent to the perspective of parents and children. Therefore, this systematic review assesses motivating and discouraging factors for children and their parents to decide to participate in clinical drug research. Studies were identified from searches in 6 databases. Two independent reviewers screened and selected relevant articles. Results were aggregated and presented by use of qualitative metasummary. 38 studies fulfilled the selection criteria and were of sufficient quality for inclusion in the qualitative metasummary. Most mentioned motivating factors for parents were: health benefit for child, altruism, trust in research, and relation to researcher. Most mentioned motivating factors for children were: personal health benefit, altruism and increasing comfort. Fear of risks, distrust in research, logistical aspects and disruption of daily life were mentioned most by parents as discouraging factors. Burden and disruption of daily life, feeling like a “guinea pig” and fear of risks were most mentioned as discouraging factors by children.

Conclusion: Paying attention to these motivating and discouraging factors of children and their parents during the recruitment and informed consent process in drug research increases the moral and instrumental value of informed consent.

What is known:

• This systematic review pools the existing empirical literature on motivations of minors and their parents to consent or dissent to participation in clinical drug research.

• The most mentioned motivating and discouraging factors for children and their parents to consent to participation in clinical drug research are identified aggregated and presented by use of qualitative metasummary.

What is new:

• This information can be used to adapt the research protocol, recruitment, and informed consent/assent process to the needs of children and their parents.

Available only for authorised users

(1998) [Dutch Medical Research (Human Subjects) Act] The Hague, the Netherlands, Staatsblad 161

Barakat LP, Patterson CA, Mondestin V, Chavez V, Austin T, Robinson MR, Li Y, Smith-Whitley K, Cohen R (2013) Initial development of a questionnaire evaluating perceived benefits and barriers to pediatric clinical trials participation. Contemp Clin Trials 34:218–226CrossRefPubMed

Baren JM, Anicetti JP, Ledesma S, Biros MH, Mahabee-Gittens M, Lewis RJ (1999) An approach to community consultation prior to initiating an emergency research study incorporating a waiver of informed consent. Acad Emerg Med 6:1210–1215CrossRefPubMed

Barrera M, D’Agostino N, Gammon J, Spencer L, Baruchel S (2005) Health-related quality of life and enrollment in phase 1 trials in children with incurable cancer. Palliat Support Care 3:191–196PubMed

Berg SL, Winick N, Ingle AM, Adamson PC, Blaney SM (2010) Reasons for participation in optional pharmacokinetic studies in children with cancer: A Children’s Oncology Group Phase 1 Consortium study. Pediatr Blood Cancer 55:119–122PubMedPubMedCentral

Brody JL, Annett RD, Scherer DG, Perryman ML, Cofrin KMW (2005) Comparisons of adolescent and parent willingness to participate in minimal and above-minimal risk pediatric asthma research protocols. J Adolesc Health 37:229–235CrossRefPubMedPubMedCentral

Brody JL, Turner CW, Annett RD, Scherer DG, Dalen J (2012) Predicting adolescent asthma research participation decisions from a structural equations model of protocol factors. J Adolesc Health 51:252–258CrossRefPubMedPubMedCentral

Broome ME, Richards DJ (2003) The influence of relationships on children’s and adolescents’ participation in research. Nurs Res 52:191–197CrossRefPubMed

Buscariollo DL, Davidson MA, Black M, Russell WE, Rothman RL, Moore DJ (2012) Factors That Influence Parental Attitudes toward Enrollment in Type 1 Diabetes Trials. PLoS One 7:e44341CrossRefPubMedPubMedCentral

10.

Cain MA, McGuinness C (2005) Patient recruitment in paediatric clinical trials. Pract Diabetes Int 22:328–332CrossRef

11.

Caldwell PHY, Butow PN, Craig JC (2003) Parents’ attitudes to children’s participation in randomized controlled trials. J Pediatr 142:554–559CrossRefPubMed

12.

Cartwright K, Mahoney L, Ayers S, Rabe H (2011) Parents’ perceptions of their infants’ participation in randomized controlled trials. J Obstet Gynecol Neonatal Nurs 40:555–565CrossRefPubMed

13.

Cherrill J, Hudson H, Cocking C, Unsworth V, Franck L, Fakis A, McIntyre J, Choonara I (2010) Clinical trials: The viewpoint of children with a chronic illness compared with healthy children. Arch Dis Child 95:229–232CrossRefPubMed

14.

Council for International Organizations of Medical Sciences (CIOMS) (2002) International Ethical Guidelines for Biomedical Research Involving Human Subjects. Geneva

15.

Critical Appraisal Skills Programme (CASP) (2014) CASP Checklists. www.casp-uk.net. Oxford, CASP

16.

Cuttini M (2000) Proxy informed consent in pediatric research: a review. Early Hum Dev 60:89–100CrossRefPubMed

17.

de Vries MC, Houtlosser M, Wit JM, Engberts DP, Bresters D, Kaspers GJ, van Leeuwen E (2011) Ethical issues at the interface of clinical care and research practice in pediatric oncology: a narrative review of parents’ and physicians’ experiences. BMC Med Ethics 12:18CrossRefPubMedPubMedCentral

18.

Deatrick JA, Angst DB, Moore C (2002) Parents’ views of their children’s participation in phase I oncology clinical trials. J Pediatr Oncol Nurs 19:114–121CrossRefPubMed

19.

Dekking SA, van der Graaf R, van Delden JJ (2014) Strengths and weaknesses of guideline approaches to safeguard voluntary informed consent of patients within a dependent relationship. BMC Med 12:52CrossRefPubMedPubMedCentral

20.

Fisher HR, McKevitt C, Boaz A (2011) Why do parents enroll their children in research: a narrative synthesis. J Med Ethics 37:544–551CrossRefPubMed

21.

Harth SC, Thong YH (1990) Sociodemographic and motivational characteristics of parents who volunteer their children for clinical research: a controlled study. BMJ 300:1372–1375CrossRefPubMedPubMedCentral

22.

Hoberman A, Shaikh N, Bhatnagar S, Haralam MA, Kearney DH, Colborn DK, Kienholz ML, Wang L, Bunker CH, Keren R, Carpenter MA, Greenfield SP, Pohl HG, Mathews R, Moxey-Mims M, Chesney RW (2013) Factors that influence parental decisions to participate in clinical research: Consenters vs nonconsenters. JAMA Pediatr 167:561–566CrossRefPubMedPubMedCentral

23.

Hoehn KS, Wernovsky G, Rychik J, Gaynor JW, Spray TL, Feudtner C, Nelson RM (2005) What factors are important to parents making decisions about neonatal research? Arch Dis Child Fetal Neonatal Ed 90:F267–F269CrossRefPubMedPubMedCentral

24.

Kimland E, Odlind V (2012) Off-label drug use in pediatric patients. Clin Pharmacol Ther 91:796–801CrossRefPubMed

25.

Koelch M, Singer H, Prestel A, Burkert J, Schulze U, Fegert JM (2009) "because I am something special" or "I think I will be something like a guinea pig": Information and assent of legal minors in clinical trials - Assessment of understanding, appreciation and reasoning. Child Adolesc Psychiatry Ment Health 3:2CrossRefPubMedPubMedCentral

26.

Lebensburger JD, Sidonio RF, Debaun MR, Safford MM, Howard TH, Scarinci IC (2013) Exploring barriers and facilitators to clinical trial enrollment in the context of sickle cell anemia and hydroxyurea. Pediatr Blood Cancer 60:1333–1337CrossRefPubMed

27.

Liaschenko J, Underwood SM (2001) Children in research: Fathers in cancer research-Meanings and reasons for participation. J Fam Nurs 7:71–91CrossRef

28.

MacNeill V, Nwokoro C, Griffiths C, Grigg J, Seale C (2013) Recruiting ethnic minority participants to a clinical trial: A qualitative study. BMJ Open 3:e002750CrossRefPubMedPubMedCentral

29.

Masiye F, Kass N, Hyder A, Ndebele P, Mfutso-Bengo J (2008) Why mothers choose to enrol their children in malaria clinical studies and the involvement of relatives in decision making: evidence from Malawi. Malawi Med J 20:50–56CrossRefPubMedPubMedCentral

30.

Menon K, Ward RE, Gaboury I, Thomas M, Joffe A, Burns K, Cook D (2012) Factors affecting consent in pediatric critical care research. Intensive Care Med 38:153–159CrossRefPubMed

31.

Miller VA, Baker JN, Leek AC, Hizlan S, Rheingold SR, Yamokoski AD, Drotar D, Kodish E (2013) Adolescent perspectives on phase I cancer research. Pediatr Blood Cancer 60:873–878CrossRefPubMedPubMedCentral

32.

Moher D, Liberati A, Tetzlaff J, Altman DG, Group P (2009) Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement. PLoS Med 6:e1000097CrossRefPubMedPubMedCentral

33.

Norris ML, Spettigue W, Buchholz A, Henderson KA, Obeid N (2010) Factors influencing research drug trials in adolescents with anorexia nervosa. Eat Disord 18:210–217CrossRefPubMed

34.

Oppenheim D, Geoerger B, Hartmann O (2005) Ethical issues in pediatric oncology phase I-II trials based on a mother’s point of view. Bull Cancer 92:E57–E60PubMed

35.

Patterson CA, Chavez V, Mondestin V, Deatrick J, Li Y, Barakat LP (2014) Clinical trial decision-making in pediatric sickle cell disease: A qualitative study of perceived benefits and barriers to participation. J Pediatr Hematol Oncol

36.

Peden V, Choonara I, Gennery B, Done H (2000) Recruiting children to a clinical trial. Paediatr Perinatol Drug Ther 4:75–78CrossRef

37.

Pinxten W (2011) One size does not fit all. Integrating ethical and legal guidance into pediatric clinical research. Catholic University Leuven, Leuven

38.

Pletsch PK, Stevens PE (2001) Children in research: Informed consent and critical factors affecting mothers. J Fam Nurs 7:50–70CrossRef

39.

Pletsch PK, Stevens PE (2001) Inclusion of children in clinical research: Lessons learned from mothers of diabetic children. Clin Nurs Res 10:140–162CrossRefPubMed

40.

Read K, Fernandez CV, Gao J, Strahlendorf C, Moghrabi A, Pentz RD, Barfield RC, Baker JN, Santor D, Weijer C, Kodish E (2009) Decision-making by adolescents and parents of children with cancer regarding health research participation. Pediatrics 124:959–965CrossRefPubMed

41.

Rothmier JD, Lasley MV, Shapiro GG (2003) Factors influencing parental consent in pediatric clinical research. Pediatrics 111:1037–1041CrossRefPubMed

42.

Sammons HM, Atkinson M, Choonara I, Stephenson T (2007) What motivates British parents to consent for research? A questionnaire study. BMC Pediatr 7:12CrossRefPubMedPubMedCentral

43.

Sandelowski M, Barroso J, Voils CI (2007) Using qualitative metasummary to synthesize qualitative and quantitative descriptive findings. Res Nurs Health 30:99–111CrossRefPubMedPubMedCentral

44.

Tait AR, Voepel-Lewis T, Malviya S (2003) Participation of children in clinical research: Factors that influence a parent’s decision to consent. Anesthesiology 99:819–825CrossRefPubMed

45.

Tait AR, Voepel-Lewis T, Siewert M, Malviya S (1998) Factors that influence parents’ decisions to consent to their child’s participation in clinical anesthesia research. Anesth Analg 86:50–53PubMed

46.

The National Commission for the Protection of Human Subjects of Biomedical and Behavioral Research (1978) The Belmont Report Ethical Principles and Guidelines for the Protection of Human Subjects of Research. U.S. Government Printing Office, Wachington D.C

47.

Truong TH, Weeks JC, Cook EF, Joffe S (2011) Altruism among participants in cancer clinical trials. Clin Trials 8:616–623CrossRefPubMed

48.

United Nations (1989) Convention on the Rights of the Child. Treaty Series 1577 (No 27531), New York

49.

US Department of Health and Human Services (2009) Protections for Children Involved as Subjects in Research (45 CFR Part 46, Subpart D)

50.

Van Stuijvenberg M, Suur MH, De Vos S, Tjiang GCH, Steyerberg EW, Derksen-Lubsen G, Moll HA (1998) Informed consent, parental awareness, and reasons for participating in a randomised controlled study. Arch Dis Child 79:120–125CrossRefPubMedPubMedCentral

51.

Vanhelst J, Hardy L, Bert D, Duhem S, Coopman S, Libersa C, Deplanque D, Gottrand F, Beghin L (2013) Effect of child health status on parents’ allowing children to participate in pediatric research. BMC Med Ethics 14:7CrossRefPubMedPubMedCentral

52.

Wagner KD, Martinez M, Joiner T (2006) Youths’ and their parents’ attitudes and experiences about participation in psychopharmacology treatment research. J Child Adolesc Psychopharmacol 16:298–307CrossRefPubMed

53.

Wendler D, Abdoler E, Wiener L, Grady C (2012) Views of adolescents and parents on pediatric research without the potential for clinical benefit. Pediatrics 130:692–699CrossRefPubMedPubMedCentral

54.

Wendler D, Shah S (2003) Should children decide whether they are enrolled in nonbeneficial research? Am J Bioeth 3:1–7CrossRefPubMed

55.

Woodgate RL, Yanofsky RA (2010) Parents’ experiences in decision making with childhood cancer clinical trials. Cancer Nurs 33:11–18CrossRefPubMed

56.

World Medical Association (2013) Declaration of Helsinki. Ethical principles for medical research involving human subjects (8th revision; original: 1964). Fortaleza

57.

Wynn L, Miller S, Faughnan L, Luo Z, Debenham E, Adix L, Fish B, Hustace T, Kelly T, MacDermott M, Marasciulo J, Martin B, McDuffie J, Murphy M, Rackoff B, Reed C, Seaman P, Thomas G, Wang W (2010) Recruitment of infants with sickle cell anemia to a Phase III trial: Data from the BABY HUG study. Contemp Clin Trials 31:558–563CrossRefPubMedPubMedCentral

58.

Zupancic JA, Gillie P, Streiner DL, Watts JL, Schmidt B (1997) Determinants of parental authorization for involvement of newborn infants in clinical trials. Pediatrics 99:E6CrossRefPubMed

Title: Motivations of children and their parents to participate in drug research: a systematic review
Authors: Krista Tromp
C. Michel Zwaan
Suzanne van de Vathorst
Publication date: 01-05-2016
Publisher: Springer Berlin Heidelberg
Published in: European Journal of Pediatrics / Issue 5/2016
Print ISSN: 0340-6199
Electronic ISSN: 1432-1076
DOI: https://doi.org/10.1007/s00431-016-2715-9

At a glance: The STEP trials

Springer Medicine

Motivations of children and their parents to participate in drug research: a systematic review

ABSTRACT

At a glance: The STEP trials

Springer Medicine

ABSTRACT

Please log in to get access to this content

Other articles of this Issue 5/2016

In vitro assessment of the effect of proportional assist ventilation on the work of breathing

CANDLE syndrome: chronic atypical neutrophilic dermatosis with lipodystrophy and elevated temperature—a rare case with a novel mutation

A quasi randomized-controlled trial to evaluate the effectiveness of clowntherapy on children’s anxiety and pain levels in emergency department

Considering behavioral interventions for comorbid psychiatric problems in Prader-Willi Syndrome: response to Bonnot et al.

Incidence of and risk factors for severe acute kidney injury in children with heart failure treated with renin–angiotensin system inhibitors

Genetic variants of ADAM17 are implicated in the pathological process of Kawasaki disease and secondary coronary artery lesions via the TGF-β/SMAD3 signaling pathway