All in the Family: Disclosure of "Unwanted" Information to an Adolescent to Benefit a Relative
Ethical assessments of clinical decisions are typically based on the preferences and interests of the individual patient. However, some clinical interventions, such as genetic testing or organ donation, may involve multiple family members. In these cases, one family member may have the potential to benefit, while another family member is exposed to potential physical or psychological risk. In the research setting, the balancing of benefits and risks between family members may be further complicated by uncertainty about their magnitude and likelihood. In addition, when the individual facing these apparently uncompensated risks is a child, the situation becomes particularly ethically complicated, as we appreciated in a recent case. Investigators at the National Cancer Institute were faced with a decision about whether it would be appropriate to disclose apparently "unwanted" research test results (length of telomeres in leukocyte subsets) to an adolescent about risk of future disease (dyskeratosis congenita), possibly causing psychological harm and an ethical wrong. These issues were not expected at the outset of the family's study participation but rather emerged with new data about the research tests. Disclosure of the research finding was an important consideration in order to avoid using the adolescent as a stem-cell donor for his sister. Disclosure to the adolescent could not be justified by merely considering the immediate interests and preferences of the adolescent. However, an expanded ethical analysis that considers the adolescent's familial context offers a more complete picture of the adolescent's interests and preferences which provides justification for disclosure.
[Show abstract] [Hide abstract] ABSTRACT: The use of predictive genetic tests is expanding rapidly. Given limited health care budgets and few national coverage decisions specifically for genetic tests, evidence of benefits and harms is a key requirement in decision making; however, assessing the benefits and harms of genetic tests raises a number of challenging issues. Frequently, evidence of medical benefits and harms is limited due to practical and ethical limitations of conducting meaningful clinical trials. Also, clinical endpoints frequently do not capture the benefit appropriately because the main purpose of many genetic tests is personal utility of knowing the test results, and costs of the tests and counseling can be insufficient indicators of the total costs of care. This study provides an overview of points to consider for the assessment of benefits and harms from genetic tests in an ethically and economically reflected manner. We discuss whether genetic tests are sufficiently exceptional to warrant exceptional methods for assessment and appraisal.
- "This consent includes " the decision in regard to the scope of the given genetic examination as well as regarding the decisions if, and if so to which extent, the examination results may be disclosed or, as the case may be, destroyed " 3 ; however, genetic information is inherited and thus shared within a family. The decision to obtain genetic information may therefore potentially impact other family members (Burnett et al. 2007; Denny et al. 2008). This is, for example, the case, if a young adult conducts a predictive genetic test for Huntington disease which had been found in a grandparent; a positive test result implies the knowledge that the parent must also be a carrier, even if he or she declined undergoing the genetic test. "
[Show abstract] [Hide abstract] ABSTRACT: Dyskeratosis congenita (DC) is an inherited bone marrow failure syndrome characterized clinically by the triad of abnormal nails, reticular skin pigmentation, and oral leukoplakia, and is associated with high risk of developing aplastic anemia, myelodysplastic syndrome, leukemia, and solid tumors. Patients have very short germline telomeres, and approximately half have mutations in one of six genes encoding proteins that maintain telomere function. Accurate diagnosis of DC is critical to ensure proper clinical management, because patients who have DC and bone marrow failure do not respond to immunosuppressive therapy and may have increased morbidity and mortality associated with hematopoietic stem cell transplantation.
- "The sibling was found to have very short telomeres prior to the discovery of the TINF2 gene mutation . The presence of very short telomeres in this potential donor was the basis of our recommendation that he not be used as the donor . Another HLA-matched sibling with normal telomeres was successfully used as the donor and was subsequently shown not to have a TINF2 mutation (which was found in the proband after the transplant). "
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