Feasibility of an Assessment Tool for Children's Competence to Consent to Predictive Genetic Testing: a Pilot Study

Abstract

Knowledge on children’s capacities to consent to medical treatment is limited. Also, age limits for asking children’s consent vary considerably between countries. Decision-making on predictive genetic testing (PGT) is especially complicated, considering the ongoing ethical debate. In order to examine just age limits for alleged competence to consent in children, we evaluated feasibility of a standardized assessment tool, and investigated cutoff ages for children’s competence to consent to PGT. We performed a pilot study, including 17 pediatric outpatients between 6 and 18 years at risk for an autosomal dominantly inherited cardiac disease, eligible for predictive genetic testing. The reference standard for competence was established by experts trained in the relevant criteria for competent decision-making. The MacArthur Competence Assessment Tool for Treatment (MacCAT-T) served as index test. Data analysis included raw agreement between competence classifications, difference in mean ages between children judged competent and judged incompetent, and estimation of cutoff ages for judgments of competence. Twelve (71 %) children were considered competent by the reference standard, and 16 (94 %) by the MacCAT-T, with an overall agreement of 76 %. The expert judgments disagreed in most cases, while the MacCAT-T judgments agreed in 65 %. Mean age of children judged incompetent was 9.3 years and of children judged competent 12.1 years (p = .035). With 90 % sensitivity, children younger than 10.0 years were judged incompetent, with 90 % specificity children older than 11.8 years were judged competent. Feasibility of the MacCAT-T in children is confirmed. Initial findings on age cutoffs are indicative for children between the age of 12 and 18 to be judged competent for involvement in the informed consent process. Future research on appropriate age-limits for children’s alleged competence to consent is needed.

Practice Variation among an International Group of Genetic Counselors on when to Offer Predictive Genetic Testing to Children at Risk of an Inherited Arrhythmia or Cardiomyopathy

“…The age at which a child develops this ability is likely variable. Hein et al suggest that children under 10 years of age are unlikely to have the competence required for involvement in the consenting process whereas children over 12 years of age are likely to be competent (Hein et al ). The European and Australian/New Zealand guidelines suggest consideration of predictive genetic testing for cardiomyopathies between 10 and 12 years of age (Charron et al ; Ingles et al ).…”

“…On the other hand, testing young children removes their autonomy and their ability to provide informed consent (Hein et al ). In addition, concerns have been raised regarding positive cardiac genetic test results potentially leading to negative psychological effects, causing overprotection and stigmatization, and resulting in discrimination later in life with regard to insurance and employability (Bonner et al ; Bratt et al ; Geelen et al ; Meulenkamp et al ; Mohammed et al ).…”

Uptake of Predictive Genetic Testing and Cardiac Evaluation for Children at Risk for an Inherited Arrhythmia or Cardiomyopathy

“…The ESC guidelines recommend waiting to pursue genetic testing for cardiomyopathies until the age at which cardiac evaluation is recommended (>10 years of age) (Charron et al 2010). This allows the child to potentially take part in the decision making process (Hein et al 2015). There is, however, uncertainty regarding a 10 year old child's ability to understand potential harms such as insurance and employment discrimination.…”

Preferences for the provision of whole genome sequencing services among young adults

“…However, the wide scope of information provided by WGS presents a major challenge for achieving fully informed decision-making and too many options may distort decision-making processes [22]. Some tools are being developed to assist in the decision-making [23,24], but the potentially serious consequences of withholding risk information makes complete reliance on the validity of patient choice controversial. For example, this concern underlies mandatory newborn screening programs [25] and recent ACMG guidelines for WGS that recommend disclosing significant unanticipated genetic risks regardless of patient preferences [26].…”