Dataset Information

Quantitative analysis of a rare disease network's international contact database and E-repository provides insights into biobanking in the electronic consent era.

ABSTRACT:

Background

Castleman disease (CD) describes a group of rare and poorly understood lymphoproliferative disorders that include unicentric CD (UCD), Human Herpes Virus-8 (HHV8)-associated multicentric CD (HHV8?+?MCD), and HHV8-negative/idiopathic MCD (iMCD). Efforts to advance research and drug discovery for CD have been slowed by challenges shared by other rare diseases, such as collecting and centralizing data and biospecimens for research. To collect disease characteristic data and identify individuals interested in contributing biospecimens for research, a global research organization - the Castleman Disease Collaborative Network (CDCN) - established an international Contact Database and electronic repository (E-repository). Herein, we performed analyses of these datasets to further characterize CD and gain insights into research biospecimen acquisition.

Results

Descriptive statistical analyses were performed on 891 participants from the Contact Database and 166 patients in the E-repository. The median age of patients at the time of enrollment in the Contact Database and E-repository was 42?±?15.7 and 35?±?14.8, respectively. The E-repository had increased representation from patients with MCD and the iMCD subtype compared to other sub-groups. Though the majority of participants were from the USA, a total of 49 countries on 6 continents were represented. Several patient characteristics in the Contact Database were associated with subsequent enrollment in the E-repository. There were significantly more MCD patients (p ConclusionsThis study of the largest- dataset of CD patients worldwide provides insights into disease phenotypes, characteristics of patients interested in contributing data and biospecimens for research, and methods for successfully acquiring data and biospecimens. Generally, the factors associated with enrollment in the E-repository represented severity of disease subtype, proximity to the research, and patient motivation. We hope that these findings and the sample documentation (e.g., electronic consent, recruitment materials) provided with this article will assist future rare disease efforts with overcoming hurdles.

SUBMITTER: Suarez A

PROVIDER: S-EPMC6625003 | biostudies-literature | 2019 Jul

REPOSITORIES: biostudies-literature

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Publications

Quantitative analysis of a rare disease network's international contact database and E-repository provides insights into biobanking in the electronic consent era.

Suarez Alexander A Reilly Curran C Fajgenbaum David C DC

Orphanet journal of rare diseases 20190711 1

<h4>Background</h4>Castleman disease (CD) describes a group of rare and poorly understood lymphoproliferative disorders that include unicentric CD (UCD), Human Herpes Virus-8 (HHV8)-associated multicentric CD (HHV8 + MCD), and HHV8-negative/idiopathic MCD (iMCD). Efforts to advance research and drug discovery for CD have been slowed by challenges shared by other rare diseases, such as collecting and centralizing data and biospecimens for research. To collect disease characteristic data and ident ...[more]

PMID: 31296233

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Project description:BACKGROUND:Efforts to improve informed consent have led to calls for providing information a reasonable person would want to have, in a way that facilitates understanding of the reasons why one might or might not want to participate. At the same time, advances in large-scale genomic research have expanded both the opportunities and the risks for participants, families, and communities. To advance the use of effective consent materials that reflect this landscape, we used empirical data to develop model consent language, as well as brief questions to assist people in thinking about their own values relative to participation. METHODS:We conducted in-person interviews to gather preliminary input on these materials from a diverse sample (n?=?32) of the general population in Nashville, Tennessee. We asked them to highlight information they found especially reassuring or concerning, their hypothetical willingness to participate, and their opinions about the values questions. RESULTS:Consent information most often highlighted as reassuring included the purpose of the biobank, the existence and composition of a multidisciplinary oversight committee, the importance of participants' privacy and efforts to protect it, and controlled access to a scientific database. Information most often highlighted as concerning included the deposition of data in a publicly accessible database, the risk of unintended access to data, the potential for non-research use of data, and use of medical record information in general. Seventy-five percent of participants indicated initial willingness to participate in the hypothetical biobank; this decreased to 66% as participants more closely considered the information over the course of the interview. A large majority rated the values questions as helpful. CONCLUSIONS:These results are consistent with other research on public perspectives on biobanking and genomic cohort studies, suggesting that our model language effectively captures commonly expressed reasons for and against participation. Our study enriches this literature by connecting specific consent form disclosures with qualitative data regarding what participants found especially reassuring or concerning and why. Interventions that facilitate individuals' closer engagement with consent information may result in participation decisions more closely aligned with their values.

Dataset Information

Quantitative analysis of a rare disease network's international contact database and E-repository provides insights into biobanking in the electronic consent era.

Background

Results

Publications

Quantitative analysis of a rare disease network's international contact database and E-repository provides insights into biobanking in the electronic consent era.

Similar Datasets

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