Skip to main content
NCBI home page
NIHPA Author Manuscripts logoLink to NIHPA Author Manuscripts
. Author manuscript; available in PMC: 2020 Sep 28.
Published in final edited form as: Hum Genet. 2017 Dec 4;137(1):63–71. doi: 10.1007/s00439-017-1855-y

Authorization of Tissues from Deceased Patients for Genetic Research

Maureen Wilson-Genderson 1, Kathryn Laura Barker 2, Heather M Gardiner 3, Maghboeba Mosavel 4, Jeffrey Thomas 5, Laura A Siminoff 6
PMCID: PMC7521139  NIHMSID: NIHMS1631647  PMID: 29204889

Abstract

Tissues from deceased donors provide important data for genomic research and Organ Procurement Organizations (OPOs) play a significant role. To understand the decisions of families who donated for transplantation and made decisions about donation to the Genotype-Tissue Expression Project (GTEx), we examined donation decisions of family decision makers (FDMs). 413 families were interviewed by telephone. The OPO staff who made the transplant and research requests completed self-administered surveys; a total of 309 matching surveys from 99 OPO staff were obtained. 76.8% of families donated to the GTEx project. Logistic regression analysis found that FDM consent to GTEx donation was associated with endorsement of policies to promote biobanking (OR=1.35), positive attitudes about medical research (OR=1.1), lack of concern regarding a breach of confidentiality (OR=1.54), comfort with tissue donation (OR=1.24), and prior authorization to solid organ donation (OR=3.17). OPO staff characteristics associated with GTEx donation included being female (OR=3.57), White (OR=4.97), comfort with hospital staff role in donation (OR=1.61), and number of topics discussed with families (OR=57.9). Donor type, FDM attitudes, OPO staff sociodemographics, OPO comfort with the GTEx authorization process, and intensity of discussing research-specific issues were significantly associated with GTEx donation decisions.

Keywords: genomic research, tissue donation, consent to biobanking, organ procurement organization

INTRODUCTION

Organ Procurement Organizations (OPOs) that obtain family authorization for deceased patient donation of organs and/or tissues for transplantation are now often asked to donate additional tissue samples for research (Bell and Leinweber 2015). This source of donated tissues for research has become increasingly important to biomedical research initiatives, especially genetic research (Ashburn et al. 2000; Baker 2012; Burke et al. 2017). Postmortem tissue donation provides several advantages to living donation, including the opportunity to obtain larger amounts of high-quality tissues, access to normal (not diseased) tissues, and a wider variety of tissue types, all of which are essential to developing treatments and increasing our understanding of disease (Mucci et al. 2013). OPOs are perhaps the single best mechanism for obtaining multiple reference tissues with high quality RNA and DNA for genomic research (Carithers et al. 2015).

The Genotype-Tissue Expression Project (GTEx)

The Genotype-Tissue Expression Project (GTEx) was funded by the National Institutes of Health’s (NIH) Common Fund to examine the relationship between gene expression and common diseases by collecting biospecimens from healthy organ and or tissue donors (Lonsdale et al. 2013; Carithers et al. 2015). The NIH partnered with six geographically dispersed OPOs to collect multiple reference tissues from over 900 donors (Lonsdale et al. 2013). These collections were accomplished via a tightly regulated protocol implemented by trained OPO staff. Potential donors were deceased individuals whose families first authorized donation for transplantation (Carithers et al. 2015). After successful completion of the pilot program in 2012, tissues were collected during a “scale-up” period between March 2013 and December 2015. Partner OPOs approached family members of deceased organ and/or tissue donors and requested an additional anatomical gift of tissues for the GTEx project. When medically suitable, families were asked to donate the whole brain (GTEx Consortium et al. 2015). Agreeing to the donation of tissues for GTEx research included authorization to release the patient’s medical records and social history, the full sequencing of the donor’s genome and, blanket consent for all future research using the donated tissue and resultant data (GTEx Consortium et al. 2015). Authorization also permits secondary researchers gaining access to the genomic data through the NIH database of Genotypes and Phenotypes (dbGaP) online data resource. The full dataset (medical, social history, and tissue) is restricted to qualified researchers who promise never to try to identify donors and must follow the standard NIH ethical guidelines for use (Keen and Moore 2015).

A separate authorization for GTEx donation was solicited from the FDM regarding the deceased’s donation for several reasons. First, authorization is nearly always sought in cases of donation of solid organ and tissues for transplantation even in the case of first-person consent by the deceased prior to death (Citation). It is true that Federal Regulations (45 CRF part 46) do not require that consent be obtained from deceased individuals (US Department of Health and Human Services 2011). However, NIH recommends that as a best practice for studies to obtain FDM authorization for donation(Office of Biorepositories and Biospecimen Research et al. 2011). This is especially poignant in the cases of genomic research wherein a breach of confidentiality could have an impact on the genomic relatives of the deceased.

Factors Affecting Authorization

Our understanding of family decision makers’ (FDMs’) tissue donation decision making, especially for research, is limited. We also lack a clear understanding of the best ethical, legal, and social policies that should guide these research projects or the types of authorization strategies currently in use (Walker et al. 2014; Grady et al. 2015; Carithers et al. 2015). Most of what we understand is focused on donation for transplantation, mostly solid organs, with a few studies of tissue donation. We do know that knowledge about tissue donation is a leading determinant in a family’s decision to donate for transplantation (Rodríguez-Villar et al. 2007; Siminoff et al. 2010; Long-Sutehall et al. 2012). In the largest study to date examining authorization to tissue donation for transplantation conducted by Siminoff and colleagues, individuals with higher incomes, more years of education, and those who were White were most likely to donate (Siminoff et al. 2010, 2011). Families are also more likely to donate for transplantation when they perceive the requester to be more caring and concerned (Siminoff et al. 2010, 2011) and when the requester is able to effectively establish rapport (Long-Sutehall et al. 2012).

The motivations of living tissue donors for research are most widely described. These donors are characterized by altruism, trust in the biobanking institution, donor control, and perceptions of benefits to self as well as others who are suffering from the same illness (Michie et al. 2011; Beskow et al. 2011; Cadigan et al. 2011; McKenzie et al. 2012; Pellegrini et al. 2012; Goodman et al. 2016). Sociodemographic variables such as age and race (Hoeyer et al. 2004; Murphy et al. 2008) are associated with authorization and it is documented that fewer minorities have participated in biobanking research than Whites (Knerr et al. 2011; Rencher and Wolf 2013). Mistrust of medical institutions and procedures may diminish enthusiasm by minority groups, who may favor more narrow consent agreements (Rencher and Wolf 2013; Ewing et al. 2015). Recent research with living donors about tissue donation for research has shown that even though minorities are under-represented in existing biobanks, members of minority communities, in the abstract, state a willingness to participate in such endeavors (Gao et al. 2014; Hagiwara et al. 2014). Reported misgivings towards living donation for genomic research and biospecimen donation include ethical, cultural, or religious concerns about the future uses of tissue samples and privacy concerns (Kaufman et al. 2009; Oliver et al. 2012; Kerath et al. 2013; Ewing et al. 2015; De Vries et al. 2016). This study collected data to understand the factors associated with FDM decision to donate their deceased family members’ tissues to a biorepository whose explicit purpose is to support future genetic research. The current study extends our previous work (Siminoff et al. 2017) and provides an examination of the characteristics of the FDMs, the tissue requesters (TRs) who asked families to donate, and the request process with decision making.

METHODS

The Ethical, Legal, and Social Issues (ELSI) Sub-Study Methods

Potential GTEx donors were newly deceased adults between the ages 21–70 whose families donated organ and/or tissue for transplantation. Donors were without significant morbidities such as cancer that would have precluded donation for transplantation.(Carithers et al. 2015) Six OPOs from geographically diverse regions of the country partnered in the parent GTEx project and assisted with tissue collection activities; five of them also participated in the GTEx ELSI sub-study. Tissue collections at all five sites were coordinated by the National Disease Research Interchange (NDRI) located in Philadelphia, Pennsylvania. NDRI provided the ELSI team with contact information for FDMs approached for GTEx donation and the donation status of each case. Details of the ELSI substudy methodology and procedures are reported elsewhere.(Siminoff et al. 2016) Virginia Commonwealth University and Temple University’s All relevant iinstitutional review boards approved this study.

The FDMs of patients who authorized organ donation were asked to donate to GTEx and were invited to participate in the ELSI sub-study, regardless of their biobanking donation decision. FDMs were mailed invitational packets two months after the patient’s death using a well-established protocol.(Siminoff et al. 2001, 2010) In the absence of an explicit refusal (opt-out card), FDMs were telephoned to further discuss the study and their potential participation. A total of 724 FDMs were invited and 413 (68.0%) provided verbal consent to participate. Semi-structured telephone interviews were subsequently held with 317 (76.8%) FDMs who donated to GTEx and with 96 (23.2%) who did not. In the case of multiple FDM, only one FDM was interviewed. The 45–75 minute interviews captured sociodemographic information, the context and content of the donation discussion, and attitudes and knowledge concerning research and biobanking. Complete details about the data collection methodology have been published elsewhere (Siminoff et al. 2016, 2017).

TRs who discussed GTEx donation with FDMs were also consented into the ELSI sub-study. TRs completed a brief online survey after each GTEx approach. The survey included questions about the GTEx authorization and the TR’s perspective on the process. Ninety-nine surveys were completed over the course of the study; each TR submitted an average of 2.8 (range 1–13) surveys.

Measurement

TRs completed demographic questionnaires upon enrollment in the study including date of birth, gender, race/ethnicity, religious preference, years working as a TR, and whether s/he held a health-related degree. We also collected FDM characteristics of gender, age, race, religion, marital status, education and income, and relationship to the patient. The primary outcome measure was the decision to donate tissues to GTEx (authorized/refused). We also recorded whether the patient donated organs or only tissues for transplantation. The latter two data points were obtained from NDRI; authorization was further confirmed during the FDM interviews.

Independent Variables

Open-ended questions queried FDMs for their reasons behind their donation decision. Individuals who reported donating only “some tissues” to GTEx while restricting others were asked both why they did and did not donate. We recorded FDMs’ concerns regarding a breach of confidentiality using the same open-ended methodology. The question gauged their general concern about any potential impact of a breach of confidentially including possible harm to themselves, genetic relatives of the donor or the donor. FDMs were classified as not at all concerned, somewhat concerned, or very concerned about the consequences of a breach of confidentiality. A single dichotomous item gauged FDMs’ willingness to donate his/her own genetic material to a biobank (yes/no). Respondents were also asked if they believed that having a signed donor card or registry participation for solid organ donation also means that the prospective donor wanted to both donate tissues and donate for research.

Seventeen items assessed biobanking-related attitudes along 5-point Likert scales with higher scores reflecting greater agreement (e.g., genetic research will improve lives). Attitudes toward medical research were assessed with a modified version of the Research Attitudes Questionnaire, which included seven items on support for medical research, trust in research, research responsibility, societal resources, research safety, research privacy, and curing diseases (Rubright et al. 2011). Item responses to the 7-point, Likert-type scale were summed to create a global attitude score, with higher values indicative of more positive attitudes toward research.

Request Process Variables

Participating TRs provided data on various aspects of the request process after each GTEx approach, including the time spent discussing GTEx with the FDM (in minutes) and the specific topics discussed. The former was solicited via a single open-ended item, while the latter was assessed using 21 dichotomous items (yes/no) representing distinct GTEx-related topics. Three 7-point Likert-type items captured TRs’ comfort discussing tissue donation for research with the family, answering the family’s questions about tissue donation for research, and comfort with healthcare workers’ roles in the authorization process; higher scores represented higher levels of comfort.

Analytic Approach

Responses to the open-ended questions were qualitatively analyzed using a systematic coding approach developed from past studies (Siminoff et al. 2010). The inter-rater reliability for the 10.5% of cases which were independently coded by two members of the research team was 99.9%; disagreements were resolved through discussion. The relationship between the decision to donate (yes/no) and FDM characteristics was assessed using the Students’ t-test statistic for continuous variables and the Chi-square statistic for categorical variables. Bivariate analyses ascertained associations between the primary outcome (authorized/refused donation) and independent and request process variables, and FDM and TR demographics. Variables with bivariate associations meeting our pre-established p <0.20 criterion were included in a subsequent binary logistic regression analysis with null information maximum likelihood method (FIML) to identify predictors of authorization. Because the sample size for TR was smaller than that for FDM, two separate logistic regression models were built to examine relationships between the independent variables and authorization. Prior to building the model, the number of topics discussed as reported by TRs transformed using proc transreg in SAS to normalize its distribution. Significance was determined at α=0.05; odds ratios (ORs) and 95% confidence intervals for the ORs are presented. Analyses were performed using IBM SPSS 21.0 for Microsoft Windows and SAS 9.4.

RESULTS

Sample Characteristics and Donation

Three hundred and seventeen (76.8%) FDMs donated to GTEx and 96 (23.2%) did not. The majority of FDMs self-identified as White, female, and Christian (Protestant or Catholic). The average age was 51.9 years, with nearly 15 years of education; just under half of the sample were widowed. Only race (White) was significantly associated with donation to GTEx, (χ2 (3, 410) = 6.6, p < .05). A strong association was found between transplantation donor type and authorizing donation for GTEx research; organ donors authorized GTEx 85% of the time, compared to tissue donors who authorized at a rate of 67.6% (χ2 (1, 412) = 17.57, p < .0001) (see Table 1).

Table 1.

Family Decision Maker (FDM) Characteristics and Donation Status (n=413)

Item Authorized n=317 (76.8%) Declined n=96 (23.2%) t-test or χ2 (p-value)
Age, mean years (SD) 52.2 (13.9) 51.0 (15.1) −0.76 (45)
Sex N (%) N (%)
 Male 91 (74.0) 32 (26.0) 0.66 (.41)
 Female 223 (77.7) 64 (22.3)
Race
 Black 42 (64.6) 23 (35.4 6.60 (.04)
 White 256 (79.3) 67 (20.7)
 Asian 19 (79.2) 5 (20.8)
 Not Reported 1 (−)
Hispanic/Latino 18 (5.7) 9 (9.4) 1.6(.20)
Marital Status 8.3(.08)
 Never Married 34 (66.6) 17 (33.3)
 Married/Cohabit 93 (82.3) 20 (17.7)
 Divorced/Separated 25 (73.5) 9 (26.5
 Widowed 150 (75.4) 49 (24.6)
Not reported 10 (100)
Religious affiliation 4.12 (0.53)
 Protestant 149 (76.8) 45 (23.2)
 Catholic 67 (73.6) 24 (26.4)
 Other 49 (84.5) 9 (15.5)
 None 52 (74.3) 18 (25.7)
Household income 3.2 (.34)
 <$30,000 45 (77.6) 13 (22.4)
 $30,000–59,999 96 (73.3) 35 (26.7)
 >$59,999 151 (80.3) 37 (19.7)
 Not reported 25 (69.4) 11 (30.6)
Education, mean years (SD) 14.8 (6.3) 14.6 (2.4) .275 (.78)
Donor Type
 Organ 183 (85.1) 32 (14.9) 17.57 (.0001)
 Tissue 134 (67.7) 64 (32.3)
Open in a new tab

Note: Values are count (percent) unless noted otherwise. Percentages may not sum to 100 due to missing values.

a

Values expressed as Mean (SD).

Similar to the samples obtained in other studies in this field,(Siminoff et al. 2010, 2015) the TR sample was predominantly White (83%) and female (73%), with an average age of 50 years. Most were Christian (61%) and either married (48%) or never married (35%). On average, TRs had 54 months’ experience and the large majority had a degree in a health-related field (81%) (data not displayed).

FDM Reasons for Donation Decision

FDMs most commonly reported positive attitudes towards donation as the reason for donating to GTEx (n=180; 61.4%). Other reasons FDMs cited were to help others (n=155, 52.9%); to support research generally (n=133, 45.4%); to support disease-focused research (n=93, 31.7%); and to support GTEx specifically (n=49, 16.7%). Noteworthy, but less cited reasons included donating because it was in line with the patient’s personality (n=20, 6.8%) or with the patient and/or family’s religious beliefs (n=7, 2.4%) (see Table 2). Apart from a few mentions of research related topics, these reasons do not vary considerably from the reasons FDMs donate for transplantation (Siminoff et al. 2007).

Table 2.

FDM Reasons for Donation Decision

Reasons for GTEx Donation n=293
 Positive towards donation in general 180 (61.4)
 Wanted to help others 155 (52.9)
 To support research 133 (45.4)
 To support disease research 93 (31.7)
 General support for GTEx 49 (16.7)
 Other 27 (9.2)
 Patient’s personality 20 (6.8)
 No response/Don’t know 10 (3.4)
 Religious beliefs 7 (2.4)
 Confuses tissue transplantation with tissue for research 7 (2.4)
Reasons Against GTEx Donation n=34
Concerns about impact on body and burial 10 (29.4)
Concerned that patient or family would not want to donate 10 (29.4)
Needed more information or time to make decision 10 (29.4)
Concerns about risks of research 7 (20.59)
Misunderstanding of GTEx request 5 (14.7)
Concerns about medical research donation 4 (11.8)
Concerns about GTEx donation 4 (11.8)
Religious Beliefs 1 (2.9)
Open in a new tab

Note: Answers sum to more than 100% due to multiple responses. Only respondents who correctly recalled their donation decision were included in this analysis.

Likewise, the reasons FDMs listed for declining to donate were rarely specifically about GTEx. The three most cited reasons (n=10, 29.4%) for declining authorization to GTEx were concerns about impact on body and burial, concerns that the patient or other family members would not want to donate, and because the FDM needed more information or time to make decision. Some FDMs did report concerns about risks of research (n=7, 20.6%), misunderstandings about some aspect of the GTEx request (n=5, 14.7%), concerns about donating to medical research (n=4, 11.8%) or to GTEx (n=4, 11.8%), and religious beliefs (n=1, 2.9%) (see Table 2). Notably, 32 nondonors (33%) incorrectly thought they actually had donated some tissues and were therefore not asked about reasons for declining donation.

FDM Donation Attitudes and Beliefs

Irrespective of donation decision, the majority of FDMs stated willingness to donate their own tissues (94.9% and 90.6%) and understood that being on a donor registry indicates willingness to donate tissues and donate for research as well as organs (81%, 70% and 79%). However, statistically significant differences were found between authorizing and refusing FDMs’ attitudes toward biobanking. For example, authorizing FDMs were less likely to report feeling squeamish and uncomfortable about tissue donation (p < .005) or to be concerned about a breach of confidentiality (p < .007). Conversely, authorizing FDMs were more likely to believe that genetic research will improve lives (p < .006) and wanted US policy to support pursuing biobanking (p < .003) (see Table 3).

Table 3.

Family Decision Maker (FDM) Knowledge and Attitudes and Donation

Item Authorized n=317 (76.8%) Declined n=96 (23.2%) t-test or χ2 (p-value)
Willing to donate own tissues (yes) 300 (94.9) 87 (90.6) 2.40 (.12)
Knows that being on a donor registry also indicates willingness to donate tissues as well as organs (% correct) 255 (81%) 76 (79%) 1.15 (0.23)
Knows that being on a donor registry also indicates willingness to donate for research as well as organs (% correct) 67 (71%) 222 (70%) .036 (.84)
Does not feel squeamish and uncomfortable about tissue donation 4.53 (1.04) 4.07 (1.38) 3.52 (.0005)
Believes genetic research will improve lives 4.84 (.46) 4.68 (.66) 2.78 (.006)
Wants US policy to support pursuing biobanking 4.69(0.79) 4.39(1.02) 3.01 (.003)
FDM not concerned about a breach of confidentiality 2.07(0.60) 1.87 (0.62) 2.70 (0.007)
Research Attitudes Scale (positive) 31.1 (3.4) 29.45 (4.5) 3.72 (.001)
Open in a new tab

TR Report of Request Process

More topics were discussed (p < .0001) and significantly more time was spent talking with FDMs (p < .03) who authorized GTEx donation than with those who declined. TRs likewise reported greater comfort discussing donation for research with FDMs who authorized research (p < .045) (see Table 4).

Table 4.

Tissue Requester (TR) Survey Data (n=309)

TR Report Authorized n=242 (76.8%) Declined n=67 (23.2%) t-test (p-value)
Comfort with discussing tissue donation for research with the family 6.0 (1.2) 5.7 (1.5)* 2.10 (.045)
Comfort with answering family’s questions about tissue donation for research 6.6 (1.2) 6.7 (1.5) 1.9 (.20)
Comfort with the role the health care providers had in the request process with this FDM 6.7 (1.2) 6.5 (1.4) 2.0 (.14)
Number of GTEx topics discussed 18.9 (3.8) 7.7 (6.5)** 16.7 (.0001)
Time spent discussing GTEx (minutes) a 22.83 (21.6) 17.15 (15.28) 2.26 (.026)
Open in a new tab

Note: Values expressed as mean (standard deviation)

Scale of 1–7; Higher values represent greater comfort. 19 possible topics

Predictors of Donation

The first logistic regression model examined relationships between the FDM characteristics, knowledge, attitudes, and authorization. The analysis revealed that authorization of GTEx donation was associated with endorsement of policies to promote biobanking (OR = 1.35), positive attitudes about medical research (OR = 1.1), lack of concern regarding a breach of confidentiality (OR = 1.54), comfort with the idea of tissue donation (OR = 1.24), and having already authorized organ, as opposed to tissue-only, donation for transplantation (OR = 3.17) (see Table 5). The second logistic regression model examined relationships between TR characteristics and their report of the request exchange and authorization. Significant predictors of donation were female gender (OR = 3.57, race (White) (OR = 4.97), comfort with other hospitals staff’s roles in the authorization process (OR = 1.61), and the number of topics discussed with FDMs (OR = 57.9) (see Table 5).

Table 5.

Logistic Regression Models for Association between Authorization to Donate Characteristics, Attitudes, and Knowledge

Odds Ratio OR Confidence Limits S.E. P-value
FDM logistic (n = 396)
Willing to donate own tissue to a biobank 0.98 (0.36–2.63) 0.503 0.967
Knows that being on a donor registry also indicates willingness to donate tissues as well as organs 1.36 (0.71–2.62) 0.333 0.351
Does not feel squeamish and uncomfortable about tissue donation- 1.24 (1.01–1.53) 0.106 0.042*
Believes genetic research will improve lives 1.23 (0.79–1.93) 0.230 0.363
Wants US policy to support pursuing biobanking 1.35 (1.02–1.78) 0.142 0.034*
FDM not concerned about a breach of confidentiality 1.54 (1.04–2.29) 0.201 0.030*
Research Attitudes Scale (positive) 1.1 (1.02–1.15) 0.032 0.014*
Donor Type (Tissue=0/ Organ = 1) 3.17 (1.85–5.43) 0.275 .0001**
Marital Status 1.15 (0.92–1.43) 0.114 0.226
Race 0.97 (0.82–1.15) 0.088 0.745
TR logistic (n = 309)
Requester age 0.99 (0.95–1.03) 0.0217 0.5700
Requester gender (Female) 3.57 (1.14–11.2) 0.5838 0.0294*
Requester race (White) 4.97 (1.34–18.5) 0.6701 0.0167*
Health-related degree (yes) 1.55 (.42–5.7) 0.6642 0.5121
Donor type (Tissue=0/ Organ = 1) 0.79 (.25–2.5) 0.5946 0.6918
Tenure as TR 1.01 (.99–1.02) 0.00584 0.1684
Comfort with the role the health care providers had in the request process with this FDM 1.61 (1.13–2.3) 0.1826 0.0091**
Comfort discussing tissue donation for research with this family 0.915 (.63–1.32) 0.1879 0.6381
Comfort answering FDM questions about tissue donation for research 0.90 (.63–1.3) 0.1871 0.5894
Number Topics discussed 57.9 (6.53–99.9) 1.1965 <.0001***
Open in a new tab
*

= p < .05,

**

p < .001

DISCUSSION

This is a novel investigation into the predictors of families’ decisions to authorize tissue donation to a genomic biobanking research project. We found that donor type (organ vs. tissue only donor), FDM attitudes, TR sociodemographic variables and comfort with the GTEx authorization process, and intensity of discussing research-specific donation issues were each significantly associated with GTEx donation decisions.

Unlike studies of the decision to donate for solid organ and tissue transplantation, FDM sociodemographic variables were not strong predictors of GTEx donation. Only race was significant in the bivariate analysis, with White FDMs being more likely to donate than FDMs of other races. TR characteristics, however, were significant predictors of authorization; specifically, women and White TRs were positively associated with donation. The lack of significant family sociodemographic characteristics is likely due to the fact that all families asked to donate to GTEx had previously agreed to donate for transplantation. These individuals are different from the general population in their willingness to perform donation behaviors.

One concern of this ‘winnowing’ of the population, is the lack of wide representation of the population at large in the tissue sample collected for research. The tissues samples were more likely to be from individuals who were White, wealthier, and more educated than the average American (Siminoff et al. 2010, 2011). Although the protocol sought take advantage of donations for therapeutic transplantation and was successful in that goal, it also resulted in a narrow demographic sample that was less representative of the population with a lower number of minorities in the research sample. The growing importance of tissues for research from deceased donors necessitates reaching out to families of eligible deceased patients who refuse organ and tissue donation for transplantation.

While families already predisposed to donate were approached about GTEx, some still declined to donate additional tissues to GTEx for research. Families who authorized GTEx donation reported their reasons for donation as focused on the purpose of the GTEx project. By contrast, nondonors’ reasons for declining were more general and underscored the need for more information. Additionally, many nondonors simply did not recall their decision, a finding supported by our pilot work (Siminoff et al. 2016, 2017). FDM who were confused about their donation decision and requested more information were directed to their OPO. Previous reports have highlighted the importance of effective communication during requests for the donation of human organs and tissue for both transplantation and research (Siminoff et al. 2001, 2010, 2011, 2015). Interestingly, donors of solid organs were more likely to donate than tissue-only donors, indicating that in-person (as opposed to telephonic) discussions were an important component of the communication context. These findings add the concern that many families never understood or actually attended to the GTEx request indicating a need to improve the request process to ensure that all families are assured of the opportunity to fully consider donation and make the most informed decision possible. Tissue requesters reported that they were able to present more topics and felt more comfortable when speaking with GTEx donor FDMs. It may be that tissue requesters need training on how to hold conversations about projects such as GTEx with individuals who are less receptive to donation.

These data also revealed that family attitudes about research were strong predictors of GTEx authorization. Donors were more likely to not concerned about potential breaches of confidentiality. We found that in the pilot assessment of risk was a significant predictor for donation with non-donors purporting more concern about the impact of a breach of confidentiality than donors and donors having a better understanding of GTEx privacy protections (Siminoff et al. 2017). This is in contrast to the findings that families with more positive attitudes toward medical research in general were more likely to donate, inclusive of beliefs in governmental support of biobanking, agreement that genetic research will improve our lives and reporting comfort with the idea of tissue donation. However, relying on a reservoir of goodwill towards research will not assist in obtaining a broader donor pool nor convince individuals who do not hold pre-existing positive attitudes toward medical research. This may be especially true as we attempt to assure that minority communities have adequate representation within biobanks to assure that the products of genomic research are applicable to the genetically diverse communities that reside in the US.

Federally funded medical research has an ethical obligation to be inclusive. Reliance on samples from homogenous populations will likely limit the applicability of the findings to individuals with genetic inheritances from Africa and Asia (Tate and Goldstein 2004). Understanding FDM attitudes regarding the donation of biological material to genomic research projects is a first step towards promoting inclusion. These findings suggest the importance of increasing opportunities to diversify the donor pool, which necessarily includes educational efforts aimed at cultivating awareness of genomic research. Familiarization with genomic tissue donation should occur well before families are presented with the option of donation during an overwhelming, highly emotional situation (i.e., the death of a family member). Concerted, culturally tailored efforts to increase genomic awareness are necessary precursors to increasing tissue donation for research purposes amongst the diverse, multicultural populations of the US.

Acknowledgements

The ELSI project would like the acknowledge the work of the GTEx OPO partners: Gift of Life in Philadelphia, PA; Center for Organ Recovery and Education in Pittsburgh, PA; LifeNet Health in Virginia Beach, VA; Washington Regional Transplant Center in Washington, DC; and LifeGift in Houston, TX. The authors would like to thank the participants for their assistance in this project.

Funding

This work was supported by the National Institutes of Health (HHSN261200800001E - Leidos Prime contract with NCI) and the National Disease Research Interchange (10XS170).

Footnotes

Conflict of Interest

On behalf of all authors, the corresponding author states that there is no conflict of interest.

Contributor Information

Maureen Wilson-Genderson, Siminoff Research Group, College of Public Health, Temple University, 1700 N. Broad Street, Suite 417, Philadelphia, PA 19121.

Kathryn Laura Barker, College of Public Health, Temple University, 1700 N. Broad Street, Suite 417, Philadelphia, PA 19121.

Heather M. Gardiner, Health Disparities Laboratory, College of Public Health, Temple University, Department of Social and Behavioral Sciences, 1301 Cecil B. Moore Ave., Ritter Annex, 9th Floor, Philadelphia, PA 19122.

Maghboeba Mosavel, Virginia Commonwealth University, Department of Health Behavior and Policy, P.O. Box: 980430, Richmond, VA 23298-0430.

Jeffrey Thomas, LifeNet Health, 1864 Concert Drive, Virginia Beach, VA 23453.

Laura A. Siminoff, College of Public Health, Temple University, 1101 W. Montgomery Ave., Philadelphia, PA 19122.

REFERENCES

  1. Ashburn TT, Wilson SK, Eisenstein BI (2000) Human Tissue Research in the Genomic Era of Medicine. Arch Intern Med 160:3377. doi: 10.1001/archinte.160.22.3377 [DOI] [PubMed] [Google Scholar]
  2. Baker M (2012) Biorepositories: Building better biobanks. Nature 486:141–6. doi: 10.1038/486141a [DOI] [PubMed] [Google Scholar]
  3. Bell TJ, Leinweber B (2015) Times Are Changing: 35 Years of Human Biospecimen Procurements for the National Disease Research Interchange. Biopreserv Biobank 13:309–310. doi: 10.1089/bio.2015.29032.tjb [DOI] [PubMed] [Google Scholar]
  4. Beskow LM, Namey EE, Cadigan RJ, et al. (2011) Research Participants’ Perspectives on Genotype-Driven Research Recruitment. J Empir Res Hum Res Ethics An Int J 6:3–20. doi: 10.1525/jer.2011.6.4.3 [DOI] [PMC free article] [PubMed] [Google Scholar]
  5. Burke GW, Posgai AL, Wasserfall CH, et al. (2017) Raising Awareness: The Need to Promote Allocation of Pancreata From Rare Nondiabetic Donors With Pancreatic Islet Autoimmunity to Type 1 Diabetes Research. Am J Transplant 17:306–307. doi: 10.1111/ajt.13983 [DOI] [PMC free article] [PubMed] [Google Scholar]
  6. Cadigan RJ, Michie M, Henderson G, et al. (2011) The meaning of genetic research results: reflections from individuals with and without a known genetic disorder. J Empir Res Hum Res Ethics 6:30–40. doi: 10.1525/jer.2011.6.4.30 [DOI] [PMC free article] [PubMed] [Google Scholar]
  7. Carithers LJ, Ardlie K, Barcus M, et al. (2015) A Novel Approach to High-Quality Postmortem Tissue Procurement: The GTEx Project. Biopreserv Biobank 13:311–319. doi: 10.1089/bio.2015.0032 [DOI] [PMC free article] [PubMed] [Google Scholar]
  8. De Vries RG, Tomlinson T, Kim HM, et al. (2016) The moral concerns of biobank donors: the effect of non-welfare interests on willingness to donate. Life Sci Soc Policy 12:3. doi: 10.1186/s40504-016-0036-4 [DOI] [PMC free article] [PubMed] [Google Scholar]
  9. Ewing AT, Erby LAH, Bollinger J, et al. (2015) Demographic Differences in Willingness to Provide Broad and Narrow Consent for Biobank Research. Biopreserv Biobank 13:98–106. doi: 10.1089/bio.2014.0032 [DOI] [PMC free article] [PubMed] [Google Scholar]
  10. Gao W, Ma GX, Tan Y, et al. (2014) Factors associated with willingness to participate in biospecimen research among Chinese Americans. Biopreserv Biobank 12:131–138. doi: 10.1089/bio.2013.0081 [DOI] [PMC free article] [PubMed] [Google Scholar]
  11. Goodman D, Johnson CO, Wenzel L, et al. (2016) Consent Issues in Genetic Research: Views of Research Participants. Public Health Genomics 19:220–8. doi: 10.1159/000447346 [DOI] [PMC free article] [PubMed] [Google Scholar]
  12. Grady C, Eckstein L, Berkman B, et al. (2015) Broad Consent for Research With Biological Samples: Workshop Conclusions. Am J Bioeth 15:34–42. doi: 10.1080/15265161.2015.1062162 [DOI] [PMC free article] [PubMed] [Google Scholar]
  13. GTEx Consortium TGte, Welter D, MacArthur J, et al. (2015) Human genomics. The Genotype-Tissue Expression (GTEx) pilot analysis: multitissue gene regulation in humans. Science 348:648–60. doi: 10.1126/science.1262110 [DOI] [PMC free article] [PubMed] [Google Scholar]
  14. Hagiwara N, Berry-Bobovski L, Francis C, et al. (2014) Unexpected Findings in the Exploration of African American Underrepresentation in Biospecimen Collection and Biobanks. J Cancer Educ 29:580–587. doi: 10.1007/s13187-013-0586-6 [DOI] [PMC free article] [PubMed] [Google Scholar]
  15. Hoeyer K, Olofsson B-O, Mjörndal T, Lynöe N (2004) Informed consent and biobanks: a population-based study of attitudes towards tissue donation for genetic research. Scand J Public Health 32:224–229. doi: 10.1080/14034940310019506 [DOI] [PubMed] [Google Scholar]
  16. Kaufman DJ, Murphy-Bollinger J, Scott J, Hudson KL (2009) Public Opinion about the Importance of Privacy in Biobank Research. Am J Hum Genet 85:643–654. doi: 10.1016/j.ajhg.2009.10.002 [DOI] [PMC free article] [PubMed] [Google Scholar]
  17. Keen J, Moore H (2015) The Genotype-Tissue Expression (GTEx) Project: Linking Clinical Data with Molecular Analysis to Advance Personalized Medicine. J Pers Med 5:22–29. doi: 10.3390/jpm5010022 [DOI] [PMC free article] [PubMed] [Google Scholar]
  18. Kerath SM, Klein G, Kern M, et al. (2013) Beliefs and attitudes towards participating in genetic research - a population based cross-sectional study. BMC Public Health 13:114. doi: 10.1186/1471-2458-13-114 [DOI] [PMC free article] [PubMed] [Google Scholar]
  19. Knerr S, Wayman D, Bonham VL (2011) Inclusion of racial and ethnic minorities in genetic research: advance the spirit by changing the rules? J Law Med Ethics 39:502–12. doi: 10.1111/j.1748-720X.2011.00617.x [DOI] [PMC free article] [PubMed] [Google Scholar]
  20. Long-Sutehall T, Winstanley E, Clarkson AJ, Sque M (2012) Evaluation of the experiences of family members whose deceased relative donated tissues at the NHSBT dedicated donation facility in Speke, Liverpool. Cell Tissue Bank 13:537–546. doi: 10.1007/s10561-011-9269-x [DOI] [PubMed] [Google Scholar]
  21. Lonsdale J, Thomas J, Salvatore M, et al. (2013) The Genotype-Tissue Expression (GTEx) project. Nat Genet 45:580–585. doi: 10.1038/ng.2653 [DOI] [PMC free article] [PubMed] [Google Scholar]
  22. McKenzie KD, Fitzpatrick PE, Sheehan JD (2012) Tissue banking: relationship with blood donor and organ donor card status. ISRN Urol 2012:475729. doi: 10.5402/2012/475729 [DOI] [PMC free article] [PubMed] [Google Scholar]
  23. Michie M, Henderson G, Garrett J, Corbie-Smith G (2011) &quot;If I Could in a Small Way Help&quot;: Motivations for and Beliefs about Sample Donation for Genetic Research. J Empir Res Hum Res Ethics An Int J 6:57–70. doi: 10.1525/jer.2011.6.2.57 [DOI] [PMC free article] [PubMed] [Google Scholar]
  24. Mucci NR, Moore HM, Brigham LE, et al. (2013) Meeting Research Needs with Postmortem Biospecimen Donation: Summary of Recommendations for Postmortem Recovery of Normal Human Biospecimens for Research. Biopreserv Biobank 11:77–82. doi: 10.1089/bio.2012.0063 [DOI] [PubMed] [Google Scholar]
  25. Murphy J, Scott J, Kaufman D, et al. (2008) Public expectations for return of results from large-cohort genetic research. Am J Bioeth 8:36–43. doi: 10.1080/15265160802513093 [DOI] [PMC free article] [PubMed] [Google Scholar]
  26. Office of Biorepositories and Biospecimen Research, National Cancer Institute, National Institutes of Health, U.S. Department of Health and Human Services (2011) NCI Best Practices for Biospecimen Resources. [Google Scholar]
  27. Oliver JM, Slashinski MJ, Wang T, et al. (2012) Balancing the risks and benefits of genomic data sharing: genome research participants’ perspectives. Public Health Genomics 15:106–14. doi: 10.1159/000334718 [DOI] [PMC free article] [PubMed] [Google Scholar]
  28. Pellegrini I, Chabannon C, Mancini J, et al. (2012) Contributing to research via biobanks: What it means to cancer patients. Heal Expect 17:523–533. doi: 10.1111/j.1369-7625.2012.00781.x [DOI] [PMC free article] [PubMed] [Google Scholar]
  29. Rencher WC, Wolf LE (2013) Ethical Research and Minority Populations Redressing Past Wrongs: Changing the Common Rule to Increase Minority Voices in Research. [DOI] [PMC free article] [PubMed]
  30. Rodríguez-Villar C, Ruiz-Jaramillo MC, Paredes D, et al. (2007) Telephone consent in tissue donation: effectiveness and efficiency in postmortem tissue generation. Transplant Proc 39:2072–5. doi: 10.1016/j.transproceed.2007.07.004 [DOI] [PubMed] [Google Scholar]
  31. Rubright JD, Cary MS, Karlawish JH, Kim SYH (2011) Measuring how people view biomedical research: Reliability and validity analysis of the Research Attitudes Questionnaire. J Empir Res Hum Res Ethics 6:63–8. doi: 10.1525/jer.2011.6.1.63 [DOI] [PMC free article] [PubMed] [Google Scholar]
  32. Siminoff L a., Traino HM, Gordon NH (2011) An Exploratory Study of Relational, Persuasive, and Nonverbal Communication in Requests for Tissue Donation. J Health Commun 16:955–975. doi: 10.1080/10810730.2011.561908 [DOI] [PMC free article] [PubMed] [Google Scholar]
  33. Siminoff LA, Gordon N, Hewlett J, et al. (2001) Factors Influencing Families’ Consent for Donation of Solid Organs for Transplantation. JAMA 286:71. doi: 10.1001/jama.286.1.71 [DOI] [PubMed] [Google Scholar]
  34. Siminoff LA, Traino HM, Genderson MW (2015) Communicating Effectively about Organ Donation: A Randomized Trial of a Behavioral Communication Intervention to Improve Discussions about Donation. Transplant direct. doi: 10.1097/TXD.0000000000000513 [DOI] [PMC free article] [PubMed] [Google Scholar]
  35. Siminoff LA, Traino HM, Gordon N (2010) Determinants of family consent to tissue donation. J Trauma 69:956–63. doi: 10.1097/TA.0b013e3181d8924b [DOI] [PMC free article] [PubMed] [Google Scholar]
  36. Siminoff LA, Traino HM, Mosavel M, et al. (2016) Family decision maker perspectives on the return of genetic results in biobanking research. Genet Med 18:82–88. doi: 10.1038/gim.2015.38 [DOI] [PMC free article] [PubMed] [Google Scholar]
  37. Siminoff LA, Wilson-Genderson M, Mosavel M, et al. (2017) Confidentiality in Biobanking Research: A Comparison of Donor and Nondonor Families’ Understanding of Risks. Genet Test Mol Biomarkers gtmb.2016.0407 doi: 10.1089/gtmb.2016.0407 [DOI] [PMC free article] [PubMed] [Google Scholar]
  38. Siminoff L, Mercer MB, Graham G, Burant C (2007) The Reasons Families Donate Organs for Transplantation: Implications for Policy and Practice. J Trauma Inj Infect Crit Care 62:969–978. doi: 10.1097/01.ta.0000205220.24003.51 [DOI] [PubMed] [Google Scholar]
  39. Tate SK, Goldstein DB (2004) Will tomorrow’s medicines work for everyone? Nat Genet 36:S34–42. doi: 10.1038/ng1437 [DOI] [PubMed] [Google Scholar]
  40. US Department of Health and Human Services: Department of Health and Human Services. Office of the Secretary. (2011) 46 CFR Parts 46, 160, and 164. Fed. Regist. 76: [Google Scholar]
  41. Walker RL, Juengst ET, Whipple W, Davis AM (2014) Genomic research with the newly dead: A crossroads for ethics and policy. J Law, Med Ethics 42:220–231. doi: 10.1111/jlme.12137 [DOI] [PMC free article] [PubMed] [Google Scholar]

RESOURCES