Pediatr Transplantation 2014: 18: 3–5
© 2014 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd
Pediatric Transplantation DOI: 10.1111/petr.12200
Editorial
Ensuring that our families are truly informed: Considerations for consent in pediatric transplantation The importance of informed consent when approaching parents in pediatric transplant settings about research, especially when medical stability has yet to be achieved, is at the heart of this issue’s paper by Dreyzin et al. (1). In this study, the authors importantly examined parent perspectives on whether or not to participate in a phase 1 hepatocyte transplant trial. Families were offered this option while waiting for a liver transplant and assured that participation would not affect their standing on the wait-list. Participants in the present study included six families who were approached about the trial (five participated and one declined). Interestingly, 10 other families declined participation and did not agree to complete this interview. Additionally, 10 families who would have been eligible while waiting for, and ultimately did receive, a liver transplant were included to determine their hypothetical considerations. Overall, the results of qualitative analyses suggested consistent areas of confusion that the authors suggest could have “adversely” affected trial participation and if addressed, could improve decision making. Perhaps a critical prerequisite to decision making from both an ethical and patient-centered standpoint is consideration of how to improve informed consent procedures under such difficult circumstances. The field of pediatric oncology contends with this situation frequently and a great deal of research reveals the complexities of this process according to different stakeholders. For example, a population-based survey administered to over 1400 parents in Germany identified from the German Childhood Cancer Registry examined parent recall of and satisfaction with the informed consent process for clinical trials (2). Data were indicative of troubling inadequacies (e.g., 14% of respondents did not
recall if they were in a trial and 3% reported incorrect recall of this; only 71% remembered providing written consent). Overall, 79% felt adequately informed. It seems unacceptable that 21% were not. Findings were exacerbated in families with one or more foreign-born parents. Of great concern, this finding has been echoed and amplified in a United States sample with researchers finding that clinicians provide less information to minority and low socioeconomic status families, which in turn leads to less engagement with the informed consent process (3). Additionally, the blur between research and clinical care indicated in this issue’s paper was highlighted here as well, in that over 50% of respondents endorsed that receiving better treatment was their motive for participating. There are a myriad of reasons why the informed consent process may become muddled that are quite relevant to pediatric transplantation. These include parent emotional stress, the clarity of research protocols, and a sense of dependency on the treatment team (4). Parents are striving to make the best decisions for their child. In an incredibly taxing context, it can become extremely difficult to distinguish between choosing to participate in research and making decisions about treatment. Clinicians recognize the anxiety faced by parents in such circumstances (5); however, the informed consent process may not adequately relieve this anxiety and confusion experienced by parents. Physicians are expected to address what Appelbaum et al. (6) coined as “therapeutic misconception,” the incorrect beliefs held by study participants that their individual needs will be considered in the research protocol and that there is a high likelihood of direct benefit from participation. However, clinicians may not sufficiently address patient misconceptions during the 3
Editorial
informed consent procedure, because of their own beliefs about the benefits of trial participation. In a study of 103 pediatric oncologists, 60% reported that patients would receive therapeutic benefit from trial participation. Even though some noted that not everyone would benefit, many clinicians still report some therapeutic optimism (5). It is likely that this hopefulness on the part of the clinician can be misconstrued by patients and their families when weighing the risks and benefits of research participation. Furthermore, the same study of pediatric oncologists found that a majority of clinicians (79%) report receiving only informal training in informed consent procedures and only 3% report that reviewing the risks and benefits to participation was their most important goal in the informed consent process (5). Without formal training, it may be difficult for clinicians to provide information to patients in a clear, culturally appropriate, and meaningful manner that clarifies misconceptions about research. Fisher (7–9) has offered a framework for achieving truly informed consent in pediatric settings. Her work must be considered here as well. She describes the importance of initiating an iterative informed consent procedure whereby investigators are sensitive and reactive to the specific needs and characteristics of the individual families. For example, in pediatric transplantation, we often see families who feel indebted to the treatment team because their child’s life has been saved. In these instances, we must be aware that such families might be eager to please when it comes to research participation. Fisher emphasizes applying strategies that go beyond aiding with decision making but to also reduce vulnerabilities inherent in the process (7–9). Some of the specific strategies that she recommends are providing age- and language-appropriate preconsent materials, encouraging families to review consent documents with trusted others before making their decision, and engaging in an active process of checking parent comprehension. Additionally, Masty and Fisher (10) provide guidance on adjusting procedures when parents are experiencing distress. For example, we often do have a sense of whether our patient’s parents are anxious or have received stressful news. It may be useful to offer families as much time as they need to consider study enrollment so that distress does not obscure their ability to gainfully participate in an informed consent meeting. This piece also helpfully discusses how, when applicable, to draw child-age patients into the discussion from a developmental perspective (10). 4
In the present study (1), the authors recommend separating discussion of trial participation from that of transplantation in order to allay confusing the two and thereby make families “more amenable” to trial participation. Separating the introduction of research procedures entirely from the clinical context may be a helpful way to alleviate some of the confusion between research and treatment (4). Finally, it may be necessary to provide formal training and/or guidelines to clinicians to help recognize and address patient vulnerabilities to truly informed consent (11). It is evident that efforts are needed to improve the informed consent process for all stakeholders, and the work of Dreyzin et al. (1) hopefully will spark a call for more attention to this process in pediatric transplantation. Research is needed that captures a broader view of what the consent process is like, especially as studies of greater complexity are launched. For example, it would be fascinating to learn from the 10 families who both chose not to participate in the hepatocyte trial and then also declined participation in the present study. Indeed, we know little about the acceptability of informed consent procedures in our field, and we cannot assume that participation rates reflect this. Rachel A. Annunziato1,2 and Melissa Rubes1,2 1 Department of Psychology, Fordham University, Bronx, NY, USA 2 Department of Pediatrics, Icahn School of Medicine at Mount Sinai, Kravis Children’s Hospital, New York, NY, USA E-mail: [email protected]
References 1. DREYZIN A, BARNATO A, SOLTYS K, et al. Parent perspectives on decisions to participate in a phase I hepatocyte transplant trial. Pediatr Transplant 2014: 18: 112–119. 2. PETERSON I, SPIX C, KAATSCH P, GRAF N, JANKA G, KOLLEK R. Parental informed consent in pediatric cancer trails: A population-based survey in Germany. Pediatr Blood Cancer 2013: 60: 446–450. 3. MILLER VA, DROTAR D, BURANT C, KODISH E. Clinician-parent communication during informed consent for pediatric leukemia trials. J Pediatr Psychol 2005: 30: 219–229. 4. DE VRIES MC, HOUTLOSSER M, WIT JM, et al. 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 2011: 27: 12–18. 5. YAP TY, YAMOKOSKI AD, HIZLAN S, et al. Informed consent for pediatric phase 1 cancer trials: Physicians’ perspectives. Cancer 2010: 116: 3244–3250. 6. APPLEBAUM PS, ROTH LH, LIDZ C. The therapeutic misconception: Informed consent in psychiatric research. Int J Law Psychiatry 1982: 5: 319–329. 7. FISHER CB. A goodness-of-fit ethic for child assent to nonbeneficial research. Am J Bioeth 2003a: 3: 27–28.
Editorial 8. FISHER CB. A goodness-of-fit ethic for informed consent to research involving persons with mental retardation and developmental disabilities. Ment Retard Dev Disabil Res Rev 2003b: 9: 27–31. 9. FISHER CB. Commentary: SES, ethnicity and goodness-of-fit in clinician-parent communication during pediatric cancer trials. J Pediatr Psychol 2005: 30: 231–234.
10. MASTY J, FISHER C. A goodness-of-fit approach to informed consent for pediatric intervention research. Ethics Behav 2008: 18: 139–160. 11. STEPAN KA, GONZALEZ AP, DORSEY VS, et al. Recommendations for enhancing clinical trials education: A review of the literature. J Cancer Educ 2011: 26: 64–71.
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Copyright of Pediatric Transplantation is the property of Wiley-Blackwell and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
© 2014 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd
Pediatric Transplantation DOI: 10.1111/petr.12200
Editorial
Ensuring that our families are truly informed: Considerations for consent in pediatric transplantation The importance of informed consent when approaching parents in pediatric transplant settings about research, especially when medical stability has yet to be achieved, is at the heart of this issue’s paper by Dreyzin et al. (1). In this study, the authors importantly examined parent perspectives on whether or not to participate in a phase 1 hepatocyte transplant trial. Families were offered this option while waiting for a liver transplant and assured that participation would not affect their standing on the wait-list. Participants in the present study included six families who were approached about the trial (five participated and one declined). Interestingly, 10 other families declined participation and did not agree to complete this interview. Additionally, 10 families who would have been eligible while waiting for, and ultimately did receive, a liver transplant were included to determine their hypothetical considerations. Overall, the results of qualitative analyses suggested consistent areas of confusion that the authors suggest could have “adversely” affected trial participation and if addressed, could improve decision making. Perhaps a critical prerequisite to decision making from both an ethical and patient-centered standpoint is consideration of how to improve informed consent procedures under such difficult circumstances. The field of pediatric oncology contends with this situation frequently and a great deal of research reveals the complexities of this process according to different stakeholders. For example, a population-based survey administered to over 1400 parents in Germany identified from the German Childhood Cancer Registry examined parent recall of and satisfaction with the informed consent process for clinical trials (2). Data were indicative of troubling inadequacies (e.g., 14% of respondents did not
recall if they were in a trial and 3% reported incorrect recall of this; only 71% remembered providing written consent). Overall, 79% felt adequately informed. It seems unacceptable that 21% were not. Findings were exacerbated in families with one or more foreign-born parents. Of great concern, this finding has been echoed and amplified in a United States sample with researchers finding that clinicians provide less information to minority and low socioeconomic status families, which in turn leads to less engagement with the informed consent process (3). Additionally, the blur between research and clinical care indicated in this issue’s paper was highlighted here as well, in that over 50% of respondents endorsed that receiving better treatment was their motive for participating. There are a myriad of reasons why the informed consent process may become muddled that are quite relevant to pediatric transplantation. These include parent emotional stress, the clarity of research protocols, and a sense of dependency on the treatment team (4). Parents are striving to make the best decisions for their child. In an incredibly taxing context, it can become extremely difficult to distinguish between choosing to participate in research and making decisions about treatment. Clinicians recognize the anxiety faced by parents in such circumstances (5); however, the informed consent process may not adequately relieve this anxiety and confusion experienced by parents. Physicians are expected to address what Appelbaum et al. (6) coined as “therapeutic misconception,” the incorrect beliefs held by study participants that their individual needs will be considered in the research protocol and that there is a high likelihood of direct benefit from participation. However, clinicians may not sufficiently address patient misconceptions during the 3
Editorial
informed consent procedure, because of their own beliefs about the benefits of trial participation. In a study of 103 pediatric oncologists, 60% reported that patients would receive therapeutic benefit from trial participation. Even though some noted that not everyone would benefit, many clinicians still report some therapeutic optimism (5). It is likely that this hopefulness on the part of the clinician can be misconstrued by patients and their families when weighing the risks and benefits of research participation. Furthermore, the same study of pediatric oncologists found that a majority of clinicians (79%) report receiving only informal training in informed consent procedures and only 3% report that reviewing the risks and benefits to participation was their most important goal in the informed consent process (5). Without formal training, it may be difficult for clinicians to provide information to patients in a clear, culturally appropriate, and meaningful manner that clarifies misconceptions about research. Fisher (7–9) has offered a framework for achieving truly informed consent in pediatric settings. Her work must be considered here as well. She describes the importance of initiating an iterative informed consent procedure whereby investigators are sensitive and reactive to the specific needs and characteristics of the individual families. For example, in pediatric transplantation, we often see families who feel indebted to the treatment team because their child’s life has been saved. In these instances, we must be aware that such families might be eager to please when it comes to research participation. Fisher emphasizes applying strategies that go beyond aiding with decision making but to also reduce vulnerabilities inherent in the process (7–9). Some of the specific strategies that she recommends are providing age- and language-appropriate preconsent materials, encouraging families to review consent documents with trusted others before making their decision, and engaging in an active process of checking parent comprehension. Additionally, Masty and Fisher (10) provide guidance on adjusting procedures when parents are experiencing distress. For example, we often do have a sense of whether our patient’s parents are anxious or have received stressful news. It may be useful to offer families as much time as they need to consider study enrollment so that distress does not obscure their ability to gainfully participate in an informed consent meeting. This piece also helpfully discusses how, when applicable, to draw child-age patients into the discussion from a developmental perspective (10). 4
In the present study (1), the authors recommend separating discussion of trial participation from that of transplantation in order to allay confusing the two and thereby make families “more amenable” to trial participation. Separating the introduction of research procedures entirely from the clinical context may be a helpful way to alleviate some of the confusion between research and treatment (4). Finally, it may be necessary to provide formal training and/or guidelines to clinicians to help recognize and address patient vulnerabilities to truly informed consent (11). It is evident that efforts are needed to improve the informed consent process for all stakeholders, and the work of Dreyzin et al. (1) hopefully will spark a call for more attention to this process in pediatric transplantation. Research is needed that captures a broader view of what the consent process is like, especially as studies of greater complexity are launched. For example, it would be fascinating to learn from the 10 families who both chose not to participate in the hepatocyte trial and then also declined participation in the present study. Indeed, we know little about the acceptability of informed consent procedures in our field, and we cannot assume that participation rates reflect this. Rachel A. Annunziato1,2 and Melissa Rubes1,2 1 Department of Psychology, Fordham University, Bronx, NY, USA 2 Department of Pediatrics, Icahn School of Medicine at Mount Sinai, Kravis Children’s Hospital, New York, NY, USA E-mail: [email protected]
References 1. DREYZIN A, BARNATO A, SOLTYS K, et al. Parent perspectives on decisions to participate in a phase I hepatocyte transplant trial. Pediatr Transplant 2014: 18: 112–119. 2. PETERSON I, SPIX C, KAATSCH P, GRAF N, JANKA G, KOLLEK R. Parental informed consent in pediatric cancer trails: A population-based survey in Germany. Pediatr Blood Cancer 2013: 60: 446–450. 3. MILLER VA, DROTAR D, BURANT C, KODISH E. Clinician-parent communication during informed consent for pediatric leukemia trials. J Pediatr Psychol 2005: 30: 219–229. 4. DE VRIES MC, HOUTLOSSER M, WIT JM, et al. 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 2011: 27: 12–18. 5. YAP TY, YAMOKOSKI AD, HIZLAN S, et al. Informed consent for pediatric phase 1 cancer trials: Physicians’ perspectives. Cancer 2010: 116: 3244–3250. 6. APPLEBAUM PS, ROTH LH, LIDZ C. The therapeutic misconception: Informed consent in psychiatric research. Int J Law Psychiatry 1982: 5: 319–329. 7. FISHER CB. A goodness-of-fit ethic for child assent to nonbeneficial research. Am J Bioeth 2003a: 3: 27–28.
Editorial 8. FISHER CB. A goodness-of-fit ethic for informed consent to research involving persons with mental retardation and developmental disabilities. Ment Retard Dev Disabil Res Rev 2003b: 9: 27–31. 9. FISHER CB. Commentary: SES, ethnicity and goodness-of-fit in clinician-parent communication during pediatric cancer trials. J Pediatr Psychol 2005: 30: 231–234.
10. MASTY J, FISHER C. A goodness-of-fit approach to informed consent for pediatric intervention research. Ethics Behav 2008: 18: 139–160. 11. STEPAN KA, GONZALEZ AP, DORSEY VS, et al. Recommendations for enhancing clinical trials education: A review of the literature. J Cancer Educ 2011: 26: 64–71.
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Copyright of Pediatric Transplantation is the property of Wiley-Blackwell and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
