Preferences in Clinical Care of Individuals With Differences of Sex Development.

OBJECTIVES: To identify the most important attributes related to the process of achieving, and outcomes associated with, successful care for differences of sex development (DSD). METHODS: We developed a best-worst scaling survey administered to 520 DSD stakeholders, including individuals or family members of those with DSD, health care specialists, and patient support and advocacy representatives. Fourteen process-related attributes and 16 outcome-related attributes were identified through qualitative research. We estimated relative importance scores and coefficients from regression analysis to understand the relative importance of attributes and conducted latent class analysis to explore heterogeneity in preferences. RESULTS: The 3 most important process attributes were (1) good communication between care team and patient/family, (2) care team educated patient/family about condition, and (3) care team incorporates the values of patient/family. The 3 most important outcome attributes were (1) patient satisfaction, (2) patient mental health, and (3) treatment maintains physical health. Latent class analyses showed that respondents had heterogeneous preferences. For process-related attributes, we identified 3 respondent groups: "Patient autonomy and support" (46% of respondents), "Education and care transitions" (18%), and "Shared decision-making" (36%). For outcome-related attributes, we identified 2 respondent groups: "Preserving function and appearance" (59% of respondents) and "Patient health and satisfaction" (41%). CONCLUSIONS: Outcomes such as patient satisfaction and health were the most important outcome attributes, and good communication and education from the care team were the most important process attributes. Respondents expressed heterogeneous preferences for selected DSD care attributes that providers should consider to improve satisfaction with and quality of DSD care.

Surgical Decision-Making for Individuals with Differences of Sex Development: Stakeholders' views.

Introduction: Advocacy and human rights organizations have called for a moratorium on elective surgical procedures until the patient is able to fully participate in the decision-making process. Due to the controversial nature surrounding surgery in differences of sex development (DSD) care, we aimed to assess the factors that teens and adults with DSD, parents, healthcare providers and other allied professionals consider pertinent to complex surgical decisions in DSD. Methods: Stakeholders (n=110) in DSD care participated in semi-structured interviews exploring features and potential determinants of successful healthcare outcomes. Audio-recordings were transcribed, coded, and analyzed using qualitative data software. Codes for "Process of Decision-Making" and "Successful Outcome-Surgery/Appearance/Function" were further searched using keywords "surgery," "procedure," and "timing." Results: Several themes were identified: 1) The nature or type of the decision being made; 2) Who should be involved in the decision-making process; 3) Timing of conversations about surgery; 4) Barriers to decision-making surrounding surgery; 5) The elements of surgical decision-making; and 6) The optimal approach to surgical decision-making. Many stakeholders believed children and adolescents with DSD should be involved in the process as developmentally appropriate. Conclusion: DSD include a wide range of diagnoses, some of which may require urogenital reconstruction to relieve obstruction, achieve continence, and/or address other anatomical differences whether cosmetic or functional. Adolescents and adults with DSD desired autonomy and to be part of the decision-making process. Parents were divided in their opinion of who should be involved in making elective surgical decisions: the child or parents as proxy medical decision-makers. Providers and other professionals stressed the importance of process and education around surgical decisions. Ongoing research examines how decision-makers evaluate tradeoffs associated with decision options.

Decisional Support Needed when Facing Tough Decisions: Survey of Parents with Children having Differences of Sex Development.

Introduction: Parents of infants and young children newly diagnosed with differences of sex development (DSD) commonly face medical and psychosocial management decisions at a time when they are first learning about the condition and cannot consult their child for input. The aim of this study was to identify areas of greatest need for parental decisional support. Methods: 34 parents of children receiving care for DSD at one of three US children's hospitals participated in a survey to learn what clinical and psychosocial decisions needed to be made on behalf of their child. Parents were then asked to identify and focus on a "tough" decision and respond to questions assessing factors affecting decision-making, decision-making preferences, decisional conflict, and decision regret. Descriptive analyses were conducted. Results: Decisions about surgery and aspects of sharing information about their child's condition with others were the two most frequently reported decisions overall, experienced by 97% and 88% of parents, as well as most frequently nominated as tough decisions. Many parents reported mild to moderate levels of decisional conflict (59%) and decision regret (74%). Almost all parents (94%) reported experiencing at least one factor as interfering with decision-making (e.g., "worried too much about choosing the 'wrong' option"). Parents universally reported a desire to be involved in decision-making - preferably making the final decision primarily on their own (79%), or together with their child's healthcare providers (21%). The majority of parents judged healthcare providers (82%) and patient/family organizations (58%) as trustworthy sources of information. Discussion: Parents of children with DSD encounter medical, surgical, and psychosocial management decisions. Despite difficulties including emotional distress and informational concerns (including gaps and overload), parents express strong desires to play key roles in decision-making on behalf of their children. Healthcare providers can help identify family-specific needs through observation and inquiry in the clinical context. Together with families, providers should focus on specific clinical management decisions and support parental involvement in making decisions on behalf of young children with DSD.

"It became easier once I knew": Stakeholder perspectives for educating children and teenagers about their difference of sex development.

OBJECTIVE: Secrecy about a child's difference of sex development (DSD) can lead to internalized shame and stigma. We explored how teenagers and adults with DSD, parents, healthcare providers, and allied professionals value and perceive patient education. METHODS: Stakeholders (n = 110) completed qualitative semi-structured interviews. Relevant themes for educational content were queried and organized. RESULTS: Education was consistently identified as essential to successful outcomes. There was less consistency in how to educate patients. Disagreement existed regarding who should champion the education process. Participants believed medically relevant information should be shared gradually with attention to developmental capacity. Details were lacking regarding how much or what information to share. Participants noted that vetted resources were helpful. Benefits of sharing condition-specific information with patients included supporting their psychosocial development. Barriers included parental resistance to sharing information due to shame/stigma, and cultural and/or family dynamics. CONCLUSIONS: Stakeholders' different perspectives regarding patient DSD education warrant future research to focus on the design, evaluation, and implementation of education-focused interventions. PRACTICE IMPLICATIONS: Healthcare providers are responsible for supporting the education of children and teenagers with DSD about their condition. When considering barriers, adopting a cultural or family systems framework can reduce parental resistance and promote open dialogue.

Fertility in Individuals with Differences in Sex Development: Provider Knowledge Assessment.

OBJECTIVE: Infertility is common among individuals with differences in sex development (DSD), and affected individuals and families desire fertility counseling. This survey sought to assess fertility knowledge and experiences with fertility counseling among DSD specialists for DSD conditions excluding congenital adrenal hyperplasia. DESIGN, SETTING, PARTICIPANTS, AND MEASURES: A survey was iteratively developed by members of the DSD-Translational Research Network (DSD-TRN) Fertility Preservation Workgroup and disseminated to 5 clinician groups: the DSD-TRN, the Society for Pediatric Psychology DSD Special Interest Group (SIG), the Pediatric Endocrine Society DSD-SIG, the Societies for Pediatric Urology, and the North American Society for Pediatric and Adolescent Gynecology. RESULTS: Completed surveys (n = 110) were mostly from pediatric urology (40.3%), gynecology (25.4%), and endocrinology (20.9%) specialists. Most (73/108, 67.6%) respondents reported discussing fertility potential. Sixty-seven responded to questions regarding fertility potential. Many participants answered questions about the presence of a uterus in individuals with 46,XY complete gonadal dysgenesis and about the potential for viable oocytes in individuals with 46,XY partial gonadal dysgenesis incorrectly. Comments acknowledged the need for further education on fertility in individuals with DSD. CONCLUSIONS: Many DSD providers have some knowledge of fertility potential, but knowledge gaps remain. Experts expressed a desire for education and accessible resources to counsel effectively about fertility potential for individuals with DSD.

Defining successful outcomes and preferences for clinical management in differences/disorders of sex development: Protocol overview and a qualitative phenomenological study of stakeholders' perspectives.

INTRODUCTION: Utilizing a qualitative phenomenological design, the Defining Successful Outcomes and Trade-offs study examined stakeholder perspectives regarding optimal healthcare delivery and outcomes for individuals with a difference/disorder of sex development (DSD). OBJECTIVE: We describe study methods and provide an overview of themes and subthemes. STUDY DESIGN: Interviews were conducted with individuals with a DSD (n = 24), parents of those with a DSD (n = 19), healthcare providers (n = 37), and others (n = 30). Primary questions regarding clinical management of patients with DSD were: "What is a successful outcome?" and "How do you achieve it?" RESULTS: Themes included: understanding of DSD diagnosis and self-efficacy in management is necessary but complex; patient and family psychological well-being; support from others versus being stigmatized; affected person experiences physical health and accepts the implications of their condition; complexities in DSD decision making, roles and expectations; and knowledgeable providers and multidisciplinary teams are essential, notwithstanding persisting barriers. Participants recognized competing values potentially forcing trade-offs in decision making. DISCUSSION: Recognition of diverse and sometimes conflicting perspectives regarding optimal pathways of care and outcomes - both within and among those with DSD and their providers -promises to enhance shared decision making. CONCLUSION: Diverse perspectives and perceptions of trade-offs associated with DSD healthcare emphasize the need to tailor care for patients and families.

Youth sports participation and health status in early adulthood: A 12-year follow-up.

Examining association between serial participation in youth organized sports (OS) and concurrent cardiometabolic risk factors, with long-term health status, will aid understanding the role of OS participation. Combining data from a prospective study and a follow-up survey, we aimed to determine association between youth OS participation and cardiometabolic risk factors with health and physical activity (PA) in young adulthood. Cardiometabolic risk factors were monitored yearly, and OS involvement through middle school, high school, and college, together with current status was recorded 12 years after the initial study began; 462 participants completed follow-up. Cardiometabolic risk factors included: body mass index (BMI), waist circumference, blood pressure, total cholesterol, high-density lipoprotein (HDL), low-density lipoprotein (LDL) cholesterol and triglycerides, fasting glucose and insulin. Participants continuing OS participation from middle to high school and/or college had significantly lower BMI/BMI z-scores [24.7 vs 27.4 (p < 0.05) and 0.51 vs 0.82 (p < 0.05), respectively] and significantly higher HDL [48.7 vs 45.4 (p ≤ 0.05)] than those without OS participation after middle school. Waist circumference of females was significantly smaller in those who participated in OS from middle to high school and/or college (84.9 cm) compared to those who had no OS (92.1 cm), (p < 0.05). Participants continuing OS from middle to high school and/or college reported significantly higher follow-up PA levels than those who had no OS after middle school (p < 0.001). This study provides novel data showing serial participation in OS from middle to high school and/or college was associated with a superior cardiometabolic health profile in school and increased PA levels at 12-year follow-up.

The National Physicians Cooperative: transforming fertility management in the cancer setting and beyond.

Once unimaginable, fertility management is now a nationally established part of cancer care in institutions, from academic centers to community hospitals to private practices. Over the last two decades, advances in medicine and reproductive science have made it possible for men, women and children to be connected with an oncofertility specialist or offered fertility preservation soon after a cancer diagnosis. The Oncofertility Consortium's National Physicians Cooperative is a large-scale effort to engage physicians across disciplines - oncology, urology, obstetrics and gynecology, reproductive endocrinology, and behavioral health - in clinical and research activities to enable significant progress in providing fertility preservation options to children and adults. Here, we review the structure and function of the National Physicians Cooperative and identify next steps.

Perceived socioeconomic status: a new type of identity that influences adolescents' self-rated health.

PURPOSE: The cognitive, social, and biological transitions of adolescence suggest that subjective perceptions of social position based on the socioeconomic hierarchy may undergo important changes during this period; yet how such perceptions develop is poorly understood, and no studies have assessed whether changes in such perceptions influence adolescents' health. This study describes adolescents' subjective perceptions of familial socioeconomic status (SSS), how SSS changes over time, and how age, race, and objective socioeconomic status (SES) indicators influence SSS. In addition, the study determines whether SSS independently influences adolescents' self-rated health, an important predictor of morbidity and health service use. METHODS: A total of 1179 non-Hispanic black and white baseline 7-12th graders from a Midwestern public school district completed a validated, teen-specific measure of SSS annually for 4 consecutive years. A parent provided information on SES. Markov modeling assessed transitions in SSS over time. RESULTS: SSS declined with age (p = .001) and stabilized among older teens. In addition to age, SES and race, but not gender, were significant correlates of SSS, but the relationships between these factors were complex. In cross-sectional and longitudinal analyses, black teens from families with low parent education had higher SSS than white teens from similarly educated families, whereas white teens from highly educated families had higher SSS than black teens from highly educated families. Lower SSS and changes in SSS predicted poor self-rated health even when adjusting for race and objective SES measures. CONCLUSION: Subjective evaluations of socioeconomic status predict adolescents' global health ratings even when adjusting for the sociodemographic factors that shape them.

Social disadvantage and adolescent stress.

PURPOSE: Low socioeconomic status (SES) and minority race/ethnicity are both associated with chronic stress and co-vary in American society. As such, these factors are often used synonymously, without clear theoretical conceptualization of their roles in the development of stress-related health disparities. This study theorized that race/ethnicity and SES reflect social disadvantage, which is the underlying factor in the development of stress-related illness, and examined how social disadvantage, defined in terms of both race/ethnicity and SES, influences adolescents' stress. METHODS: This is a cross-sectional school-based study of 1209 non-Hispanic black and white 7th-12th graders from a single Midwestern metropolitan public school district. Each student completed a questionnaire and a parent provided SES information. Race/ethnicity was obtained from school records. Linear regression analyses determined the influence of race/ethnicity and SES to stress. Race/ethnicity and presence or absence of at least one parent who graduated from college were used to define four subgroups for within-group analyses. RESULTS: Stress was higher among black students, those from lower SES families, and those with lower perceived SES. In subgroup analyses, neither race nor SES maintained their independent associations with stress among socially disadvantaged groups. Black race was not associated with stress among those without a college-educated parent, and parent education did not influence stress among black students. In contrast, among more socially advantaged groups, both SES and race explained variation in adolescents' stress. CONCLUSIONS: Social disadvantage is associated with increased stress, regardless of whether disadvantage is defined in terms of race or SES. This suggests that race and SES measure adversity in the social environment, and therefore, serve as risk markers, rather than risk factors. Future research should focus on the experience of adversity, which is reflected by these social characteristics, and the processes by which it operates.