Ellen S. Tambor

Elementary school-aged children's reports of their health: A cognitive interviewing study

There are no standard methods for assessing the quality of young childrens perceptions of their health and well-being and their ability to comprehend the tasks involved in reporting their health. This research involved three cross-sectional studies using cognitive interviews of 5–11-year-old children (N = 114) to determine their ability to respond to various presentations of pictorially illustrated questions about their health. The samples had a predominance of children in the 5–7-year-old range and families of lower and middle socio-economic status. The research questions in Study 1 involved childrens ability to convert their health experiences into scaled responses and relate them to illustrated items (n = 35); Study 2 focused on the type of response format most effectively used by children (n = 19); and Study 3 involved testing childrens understanding of health-related terms and use of a specific recall period (n = 60). The results of Study 1 showed that children identified with the cartoon drawing of a child depicted in the illustrated items, typically responding that the child was at or near their own age and of the same gender, with no differences related to race. Study 2 results indicated that children responded effectively to circles of graduated sizes to indicate their response and preferred them to same-size circles or a visual analogue scale. Tests of three-, four-, and five-point response formats demonstrated that children could use them all without confusion. In Study 3, expected age-related differences in understanding were obtained. In fact, the 5-year-old children were unable to understand a sufficient number of items to adequately describe their health. Virtually all children 8 years of age and older were able to fully understand the key terms and presentation of items, used the full five-point range of response options, and accurately used a 4-week recall period. Six- and seven-year-olds were more likely than older children to use only the extreme and middle responses on a five-point scale. No pattern of gender differences in understanding or in use of response options was found. We conclude that children as young as eight are able to report on all aspects of their health experiences and can use a five-point response format. Children aged 6–7 had difficulty with some health-related terms and tended to use extreme responses, but they understood the basic task requirements and were able to report on their health experiences. These results provide the guidance needed to develop and test a pediatric health status questionnaire for children 6–11 years old.

Improving response rates through incentive and follow-up: The effect on a survey of physicians' knowledge of genetics

OBJECTIVES This study assessed efforts to increase response rates to a mailed physician survey and examined whether, as a result, nonresponse bias was reduced. METHODS Randomly selected physicians and geneticists were mailed a questionnaire concerning genetics knowledge and attitudes. In the final but not the pilot survey, a

Informed consent for enrolling minors in genetic susceptibility research: a qualitative study of at-risk children’s and parents’ views about children’s role in decision-making

25 incentive and intensive follow-up were used to increase the response rate. RESULTS The response rate from physicians in the final survey was 64.8% (n = 1140), compared with 19.6% in the pilot test (n = 69). Sample representatives in sociodemographic and practice characteristics was improved by follow-up. Respondents recruited with more difficulty did not differ on the principal outcome variable, genetics knowledge, except on one subscore. Pilot study and final survey respondents did not differ in knowledge. CONCLUSIONS Although the effect of increased response rates on the principal outcome variable in this study was minimal, this may not be the case for other studies. Every effort should be made to attain as high a response rate as is practical and to establish that respondents are representative of the population being sampled.

Mapping the human genome: an assessment of media coverage and public reaction.

PURPOSE To better understand the process by which families at increased risk of disease would decide to enroll their children in genetic susceptibility research in order to develop recommendations regarding the informed consent process by which at-risk children are enrolled in such research in the future [corrected]. METHODS Parents and children (ages 10-17 years) from families at increased risk for heart disease (n = 21 dyads) or breast cancer (n = 16 dyads) participated in two face-to-face, audio-taped, semi-structured interviews: Initial interviews were conducted with parents and children separately, and follow-up family interviews were conducted 1 year later. Interview transcripts were coded based on common themes. RESULTS Families vary in the stage at which, and degree to which, children would be involved in decision-making about research participation. In general, the older/more mature the child, the less risky the research and the more open the communication style, the greater the likelihood that decisions would be made jointly. Most children wanted some parental input, but still thought the final decision should be theirs. Most parents would want to make the initial decision about whether it would be reasonable to consider enrolling their child in the research being proposed, but none opposed the child having some time alone with the researcher. All parents and children in our study placed extreme importance on not forcing children to participate in nontherapeutic research if they do not want to. CONCLUSIONS Decision-making about enrolling children in genetic susceptibility research should be based on an informed consent process that (a) gives parents and children sufficient opportunity to ask questions of the researcher(s) and to communicate with one another, and (b) gives children the opportunity to exercise their right to refuse participation without parental influence. This process should be tailored to the childs maturity level and style of communication in the family.

The quality of media reports on discoveries related to human genetic diseases.

Purpose: To assess public reactions to the June 26, 2000, announcement that scientists had nearly finished mapping the human genome.Methods: We conducted a random-digit telephone survey of 407 Maryland residents as well as a content analysis of 55 relevant media reports.Results: African Americans were more likely than Caucasians to report a negative reaction (P < 0.001) to the genome announcement. Overall, privacy/discrimination (16%) and human cloning (14%) were the most commonly mentioned concerns regarding the impact of the genome mapping.Conclusions: These findings highlight the need for continued public discourse, including through the media, to address concerns regarding the Human Genome Project.

Physicians'Attitudes toward Disclosure of Genetic Information to Third Parties

Objectives: To examine (1) the quality of media reports (newspapers, television and public radio) of genetic discoveries with medical relevance and (2) factors related to the completeness and balance of the stories. Methods: Analysis of the accuracy, balance, and completeness of 228 media stories reporting 24 genetic discoveries between 1996 and 2000 using a previously validated instrument. Results: Although usually accurate, the stories contained only 45.5 ± 13.8% (mean ± SD) of relevant items. Stories appearing on television and stories reporting discoveries of genes for rare diseases were the least complete. Stories in non-US English-speaking newspapers included more content items per word than US stories. Less balanced stories exaggerated the benefits of discoveries, ignored possible risks, and did not present a range of expert opinion. Scientists were sometimes the source of exaggeration. Conclusions: To increase the quality of media reports about genetic discoveries, stories should include more relevant items and be written by journalists skilled in science writing. Scientists will have to resist the tendency to exaggerate. These conclusions may apply to media stories of other discoveries as well.

Should Women at Increased Risk for Breast and Ovarian Cancer Be Randomized to Prophylactic Surgery? An Ethical and Empirical Assessment

onfidentiality is a cornerstone of the physicianpatient relationship. Breaches of confidentiality C in the context of genetic testing are of particular concern for a number of reasons.’ First, genetic testing reveals information not only about a particular patient, but also about his or her family members. Second,genetic testing can label healthy people as “at risk,” subjecting them to possible stigmatization or discrimination by third parties. Third, as genetic testing becomes more widespread and is incorporated into primary care, breaches of confidentiality might inadvertently occur more frequently because primary care providers may not be trained to understand the uniqueness of genetic information. Until now, genetic services have been provided primarily by medical geneticists and genetic counselors. However, with the proliferation of new genetic presymptomatic and carrier tests, primary care physicians are going to become increasingly involved in genetic testing2 Currently, little is known about physicians’ attitudes (other than those of medical geneticists3 ) toward disclosure of confidential genetic information to third parties.

Stakeholder perspectives regarding alternate approaches to informed consent for comparative effectiveness research

More information is needed about the relative effectiveness of prophylactic surgery, chemoprevention, and surveillance in reducing breast and ovarian cancer risk in women with an inherited susceptibility mutation. We assessed practical and ethical barriers to conducting randomized clinical trials (RCTs) to compare preventive interventions for breast and ovarian cancer. Eighty-seven at-risk women who attended an education and counseling session about BRCA1/2 testing were asked about their willingness to participate in hypothetical research studies for breast and ovarian cancer risk reduction. In addition, 247 Maryland physicians from five specialties completed a mail survey including a question about their likelihood of recommending RCT participation to an at-risk woman. Nineteen percent of at-risk women reported willingness to participate in a hypothetical RCT for breast cancer risk reduction and 17% for ovarian cancer risk reduction. Women with children and women likely to have a prophylactic mastectomy if found to have a susceptibility mutation were significantly more willing to participate in an RCT. A majority of women would be willing to participate in nonrandomized trials or registries. Fifty-two percent of physicians responded that they would be likely to recommend RCT participation to a woman carrying a breast cancer susceptibility mutation. Oncologists were the most likely to recommend an RCT. Although the results of nonrandomized trials may be difficult to interpret because of such issues as selection bias. Greater feasibility combined with fewer ethical concerns make nonrandomized trials a more viable alternative to randomized trials for evaluation of preventive interventions for breast and ovarian cancer when prophylactic surgery is one of the treatments being evaluated.

Self-Esteem as an Interpersonal Monitor: The Sociometer Hypothesis

Traditional informed consent approaches, involving separate discussions and lengthy consent forms, may be an imperfect fit for comparative effectiveness research (CER) that is integrated into usual care and compares non‐investigational treatments. However, systematic efforts to collect broad stakeholder perspectives about alternative streamlined approaches to disclosure and consent in this context have been limited.