Annie G Steinberg’s research while affiliated with The Children's Hospital of Philadelphia and other places

Few studies examine whether and how parental attitudes towards genetic testing change over time. In this study we interviewed parents of 14 children with newly identified hearing loss at two time points: after referral to genetics and 1 year later. Qualitative analyses of parental narratives indicate that parental attitudes did not change significantly over this time. Parents who perceived genetic testing to be useful continued to value it after testing, while parents who did not perceive it as being useful for their child's future held the same view a year later. The only parents who changed their views regarding the usefulness of genetic testing for hearing loss were those who reported that their children underwent significant changes in their hearing loss or were faced with other life threatening conditions. Parents were also often unaware of the role of the genetic counselor and how genetic counseling could help address many of their lingering questions and concerns. These emergent themes indicate the need for geneticists and genetic counselors to be aware of and sensitized to the questions and attitudes that bring parents to a genetic evaluation, as well as the reasons why parents may not follow up with genetic testing for hearing loss when recommended.

We report on the development of a Genetic Attitude Assessment Tool (GAAT) to measure parental attitudes in contemplating genetic testing for childhood hearing loss, and to examine the differences in assessments made before and after genetic counseling. The GAAT tool was administered to a convenient sample of 119 parents of children with bilateral sensorineural hearing loss. The respondents completed the survey either before (n = 77) or after (n = 42) genetic counseling. Exploratory Factor Analysis was applied to identify and quantify the underlying psychosocial structure. Our results showed the validated 54-item GAAT instrument contains six subscales: (1) "test intention," (2) "beliefs in non-genetic causes of hearing loss," (3) "deferral of decision to undergo genetic testing," (4) "appropriate use of genetic testing results," (5) "beliefs in the benefits," and (6) "concerns about stigma." The respondents who answered the survey after genetic counseling had higher "test intention" (P = 0.017) and endorsed to a greater extent "beliefs in the benefits" (P < 0.001). They believed to a lesser extent that childhood hearing loss was due to "non-genetic causes" (P < 0.001) and were less inclined to prefer "decision deferral" (P = 0.031). Respondents who themselves had a hearing loss expressed a significantly weaker belief in "non-genetic causes" of hearing loss (P < 0.0001). In conclusion the validated GAAT instrument is responsive to changes in parental attitudes after genetic counseling. The GAAT may be used to monitor parental attitudes serially, to further understand how parental attitudes change from pre genetic counseling, post genetic counseling, to post test result disclosure.

Studies on parental attitudes towards genetic testing for hearing loss have surveyed parents of newborns with hearing loss as well as deaf and hearing adults. Although research indicates that most people have positive attitudes about genetic testing, few studies examine parental narratives about the personal implications of genetic hearing loss in their children. In this qualitative study we conducted semi-structured interviews with 24 parents whose children had been referred for, but had not yet undergone, genetic testing for hearing loss. The parents were recruited to represent a diverse range of racial, ethnic, and socioeconomic groups. Genetics and genetic testing for hearing loss were poorly understood topics. Beyond supporting or opposing genetic testing for hearing loss, parents' construction of meaning included struggles to locate responsibility (metaphysical attributions, ascription or alleviation of parental responsibility) as well as questions about the usefulness and implications of genetic testing for hearing loss in their child. Based on the themes that emerged from this study, we highlight the need for healthcare professionals to be aware and sensitized to parents' narratives, personal meanings and socio-cultural context when referring them for genetic testing for hearing loss. Listening attentively to parental narratives can help minimize prevailing misconceptions among parents and enable appropriate medical care and education.

Purpose Few studies have examined how parents personalize the possibility of genetic hearing loss in their children and whether they actually intend to pursue testing for their child. This article addresses the audiologist’s important role in the genetic testing referral and follow-up processes. Method Twenty-four parents whose children were referred to genetic testing for hearing loss were interviewed in depth. Parents were selected to include a diverse range of races, ethnicities, and socioeconomic levels. Interviews were coded and analyzed using qualitative methods. Results Parental associations with genetic testing included feeling personally responsible, feeling relief, and considering metaphysical attributions for their child’s hearing loss. Parental attitudes were related to perceptions and experiences with deafness. Many misconceptions about genetics were also found. Conclusions Audiologists need to be sensitized to parents' personal and sociocultural contexts when discussing genetic testing and should tailor informational and emotional support to parents' requirements when confronting the possibility of their child having a genetic hearing loss.

People who are deaf use health care services differently than the general population; little research has been carried out to understand the reasons. To better understand the health care experiences of deaf people who communicate in American Sign Language. Qualitative analyses of focus group discussions in 3 U.S. cities. Ninety-one deaf adults who communicate primarily in American Sign Language. We collected information about health care communication and perceptions of clinicians' attitudes. We elicited stories of both positive and negative encounters, as well as recommendations for improving health care. Communication difficulties were ubiquitous. Fear, mistrust, and frustration were prominent in participants' descriptions of health care encounters. Positive experiences were characterized by the presence of medically experienced certified interpreters, health care practitioners with sign language skills, and practitioners who made an effort to improve communication. Many participants acknowledged limited knowledge of their legal rights and did not advocate for themselves. Some participants believed that health care practitioners should learn more about sociocultural aspects of deafness. Deaf people report difficulties using health care services. Physicians can facilitate change to improve this. Future research should explore the perspective of clinicians when working with deaf people, ways to improve communication, and the impact of programs that teach deaf people self-advocacy skills and about their legal rights.

This paper will describe the genesis and philosophy of the Center for Children's Policy. Practice and Research at the University of Pennsylvania (CCPPR), a joint project of the Schools of Law, Medicine and Social Work. The mission of CCPPR was to mobilize the resources of all disciplines engaged in childhood issues across the campus to seek innovative solutions for the crises facing America's children. CCPPR seeks to combine clinical practice, research, and policy in a vertical structure that would maintain linkages at all stages of scholarship, practice, and reform. Five essential structural factors were that the project must be: (a) vertically integrated; (b) interdisciplinary; (c) team-based; (d) child-centered and (e) developmentally informed. This paper can serve as a template for developing similar centers or projects, and will describe our methods, the projects we have undertaken, as well as the pitfalls and challenges of this highly demanding integrative approach.

Advances in cochlear implant (CI) technology have increased the complexity of treating childhood deafness. We compare parental decision-making, values, beliefs, and preferences between parents of eligible and ineligible children in considering cochlear implants. Surveys were obtained from 83 hearing parents of deaf children. A subset of 50 parents also underwent semi-structured interviews. Nine hypothetical outcomes, ranging from mainstream success to poor mainstream outcome were created to measure parents' overall preferences and preference for specific outcomes for their child who is deaf. Among parents of eligible children (n = 50), approximately 2/3 considered implantation (n = 33). The other 1/3 did not consider implantation. Parents who were eligible but did not consider implantation placed significantly lower priority on mainstream success over bilingual success (P < 0.03), and on the child's ability to speak versus sign (P < 0.02). They also showed significantly higher concerns on the cost of services in general and on the availability of resources offered at the local school district (both P > 0.05). Parents of ineligible children (n = 30) rarely considered implantation, even if they showed similar aspirations in mainstream outcomes (P = 0.003). Semi-structured interview data supported these findings. The decision to consider cochlear implantation is strongly influenced by the eligibility and by professionals' recommendations. However, for some parents, the decision goes beyond eligibility and is determined by parental preferences, goals, values, and beliefs. This highlights the importance of careful audiologic evaluation and professionals' awareness of and sensitivity to parental goals, values, and beliefs in evaluating the child's candidacy.

This chapter summarizes the findings of the research reported in preceding chapters and suggests the implications of these results for parents and educators. One important lesson to be learned from the infants participating in this study is the diversity of their experiences in the first weeks and months of life. They were born into relatively privileged environments: middle class, college-educated, two-parent families. They have the advantages of caring, nurturing parents, early diagnosis and prompt intervention, all of which combine to predict positive developmental outcomes despite the risk factors associated with their early histories. Nevertheless, their medical biographies, particular temperaments, and motor capabilities require individually designed programs for remediating existing difficulties and preventing future complications.

This chapter examines mother-infant interactions at 12 and 18 months. It shows that many deaf mothers with hearing children and hearing mothers with deaf children exhibited appropriate interactive behaviors and reinforced their child's positive responses. However, the striking group differences in mean scores between the matched hearing status dyads and those for the unmatched hearing status dyads raise a flag of concern. Hearing mothers with deaf infants routinely receive support services, mandated by federal law. This research suggests that deaf mothers with hearing infants could well benefit from similar services to help them deal with the practical and the emotional challenges of parenting a child whose communicative needs and abilities differ significantly from their own.