Ethical, Legal, and Social Issues (ELSI) of Responsible Data Sharing Involving Children in Genomics: A Systematic Literature Review of Reasons

Figures & data

Figure 1. PRISMA chart.

Table 1. Publication characteristics

	No. (%) of publications N= 151
Publication Type
Peer reviewed article	125 (82)
Guideline/policy	9 (5.9)
Book section	3 (1.9)
Report	4 (2.6)
Journal published letters	4 (2.6)
Press release	4 (2.6)
Conference proceedings	2 (1.3)
Content Type
Commentary or opinion piece	70 (46)
Empirical study	43 (28)
Data sharing SOP	27 (18)
Data sharing guideline/policy	4 (3.3)
International convention	3 (3.3)
Professional guideline	1 (0.6)
Regulation	3 (1.3)
Journal field for 139 empirical studies, commentaries and reports
Genetics/genomics	42 (30)
Medicine	40 (27)
Bioethics	17 (12)
General science	12 (8)
Public Health	10 (7.2)
Social science	6 (4.3)
Health policy/law	6 (4.3)
Informatics	4 (3)
Nursing	2 (1.4)
Region of publication or policy/guideline/law
North America	93
Europe	43
Africa	4
Other (international)	4
Asia	3
New Zealand/Australia	3
Middle East	1

Table 2. Reasons and subreasons given for why children’s genomic and associated clinical data should (n = 3) and should not (n = 149) be shared [number of mentions].

1. Genomic data privacy protection [1]  i. Potential for data misuse [1]  ii. Lack of compelling need to maintain genotypic information [1]

2. Enhance the prospect of direct and indirect benefits to individual, as well as other pediatric patients [27]  i. Direct clinical benefits for the patient [7]  ii. Indirect benefits to other, as well as future children [12]   • Indirect benefits justified on the basis of solidarity [1]  iii. Direct and indirect benefits to patient(s) [8] 3. Maximize the clinical and scientific utility of children’s data [19]  i. To improve statistical power and sample size [14] 4. Encourage research collaboration [13] 5. Advance pediatric medicine generally [12]  i. To improve diagnostic yields [4]  ii. To develop new drug therapies [5] 6. Fulfill a duty to share [8] 7. Support the learning health care system [1] 8. Prevent data waste [5] 9. To mobilize public health responses [3] 10. Enable verification and reproducibility of genomic results [6] 11. Minimize patient burdens and risks by not duplicating data collection [4]

Table 3. Reasons and subreasons given for the conditions that support “responsible” sharing of children’s genomic and associated clinical data³ [number of mentions] (Sofaer and Strech 2011).

1. Consent [29]  i. Right to recontact/reconsent for continued data use at the age of majority [35]  ii. Broad consent [11]   • Scope of parental authority to provide broad consent [29]   • Joint parental consent [2]   • Posthumous consent [1]  iii. Children’s assent and shared decision making [27]   • Familial implications of data sharing [5]  iv. Consent waivers [8]   • Ethics committee to determine recontact responsibility [7]   • Opt-in/-out for recontact [12] 2. Responsible data protection [18]  i. Data deidentification [25]  ii. Compliance with applicable laws [14]   • Technical strategies for data deidentification to comply with ethical–legal guidelines [10]   • Data linkage and implications for reidentifiability [19]  iii. Special protections for genomic data [9] 3. Balance benefits with informational risks [17]  i. Risks related to loss of privacy [12]   • Via third-party access [15]   • Via genetic discrimination [3]   • Uncertain risks via secondary/future use [4]  ii. Evaluation based on current available evidence [5]   • Evidencing the minimal risk standard [12]  iii. Physicians and investigators responsible for balancing benefits and risks [1] 4. Responsible data management [9]  i. Via interoperability and ease of use [13]   • Need for a shared lexicon for data interoperability [4]  ii. Role of institutions in data management [5]   • Need for harmonized norms [6]  iii. Via mechanisms of mutual accountability for data sharers and users [3]  iv. Clarification of data ownership [7] 5. Responsible data access [10]  i. Via data use(r) restrictions [29] 6. Sustaining patient willingness and trust [10]  i. Methods to enhance willingness and trust to share [7] 7. Continuing education and stakeholder outreach [13] 8. Incentivization and appropriate attribution for data sharers [6]

Sofaer, N., and D. Strech. 2011. Reasons why post-trial access to trial drugs should, or need not be ensured to research participants: A systematic review. Public Health Ethics 4 (2):160–184. doi: 10.1093/phe/phr013.

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