Satisfaction with health care services in young people with cerebral palsy in the transition period: results from a European multicenter study.

Background: Young people with chronic health conditions and disabilities rely on the healthcare system to maintain their best possible health. The appropriate delivery and utilization of healthcare services are key to improve their autonomy, self-efficacy and employment outcomes. The research question of our study is directed toward investigating if poor availability and accessibility of healthcare services in general, as identified by unmet needs in healthcare, are associated with dissatisfaction with healthcare. Methods: Within a European multicenter observational study, 357 young adults with cerebral palsy aged 19-28 were included. We assessed special healthcare needs, utilization of healthcare services, and satisfaction with healthcare applying the short-form of the YHC-SUN-SF, environmental and social variables (EAEQ) as well as indicators for severity of condition and functionality (e.g., GMFCS) of these participants based on a self-, assisted self- or proxy-reports. We used correlation analyses to explore associations between satisfaction with healthcare and respective indicators related to availability and accessibility of healthcare services as well as severity of the condition. In addition, we included reference values for satisfaction with heath care from young adults with various chronic conditions assessed within population-based surveys from some of the European countries included in the study. Results: We identified several unmet healthcare needs, especially for widely used and established services (e.g., physical therapy). Satisfaction with healthcare (YHC-SUN-SF general and subscale scores) was moderate to high and almost consistently better for the sample of young adults with cerebral palsy as compared to reference values for young adults with various chronic conditions assessed within general population surveys). Correlation coefficients between satisfaction with healthcare and utilization of services and (unmet) healthcare needs were low, also with different indicators for severity of the condition or functionality. Conclusion: Young adults with cerebral palsy reports of unmet healthcare needs varied largely but showed substantial deficits in some aspects. This seems to have no impact on the satisfaction with healthcare those patients currently receive. We conclude that these are two different constructs and somewhat independent indicators to evaluate the quality of healthcare. Clinicians and other practitioners should consider this distinction when monitoring patient needs in their daily practice.

Transition competence as an indicator of health outcomes related to transition.

BACKGROUND: Adolescents and young adults (AYAs) with chronic conditions face a transfer, defined as an actual shift from paediatric to adult-oriented health care. Transition competence as the self-perceived knowledge, skills and abilities regarding the transition process was considered extremely useful. AIM: This study was designed to investigate the impact of transition competence before and after the transfer on disease-specific quality of life (QoL) and health care satisfaction of AYAs with diabetes. RESULTS: In total, a sample of N = 90 AYAs with diabetes self-reported their transition competence, diabetes-specific QoL and satisfaction with care. Multiple linear regressions were used to analyse the impact of transition competence on satisfaction with care and QoL. Transition competence positively influenced the outcomes of satisfaction with care and QoL. CONCLUSION: Young adults with diabetes showed higher transition competence scores than adolescents with diabetes.

[The Relationship between Social and Family Structures and Behavioral Problems in Preschool Children]. / Der Zusammenhang von sozialen und familiären Strukturen mit Verhaltensauffälligkeiten bei Vorschulkindern.

BACKGROUND: Behavioral problems in children are indicators of compromised mental health. Their development is influenced by family and social factors, with limited understanding of interactions among family structure, educational status, migration background, and parental employment concerning behavioral problems. These associations were investigated in children between 5-6 years of age. METHODS: Data (n=15,271) were collected between 09/2018 and 08/2019 in the federal state of Schleswig-Holstein as part of school entry examinations. Children's behavioral problems were assessed using the Strengths and Difficulties Questionnaire (SDQ). Statistical analyses included descriptive assessments and the calculation of a bivariate logistic regression model, with SDQ's outcomes "problematic" and "non-problematic" as dependent variables, and social and family factors as independent variables (gender, family structure, number of siblings, language proficiency, migration background, parental education level, parental employment, and sports participation in a sports club). RESULTS: Logistic regression analyses revealed that children living with single parents had a 2.1-fold (odds ratio; OR) higher likelihood [95% confidence interval (CI): 1.7-2.6] of displaying behavioral problems compared to children living with both biological parents. Only children had a 1.4-fold higher likelihood [95% CI: 1.2-1.8] compared to children with one or two siblings. Children with a unilateral migration background exhibited a lower likelihood of behavioral problems [OR: 0.58; 95% CI: 0.38-0.87] than children without a migration background. Children from families with low educational attainment had a 3-fold higher likelihood of behavioral problems [95% CI: 2.3-3.8] compared to those from high educational attainment families. When both parents (or the single parent) were employed at least part-time, there was a lower likelihood of behavioral problems [OR: 0.58; 95% CI: 0.47-0.71] in the child compared to situations where at least one parent was unemployed. CONCLUSIONS: This study identified sociodemographic factors associated with manifestation of behavioral problems. Particularly, single parents, families with lower educational levels and families with at least one unemployed parent should be targeted for intervention.

Differences in participation between young adults with cerebral palsy and their peers: A cross-sectional multicentre European study.

BACKGROUND: Children and adolescents with disabilities are known to participate less in most areas of life than their non-disabled peers. OBJECTIVES: (1) To estimate differences in participation between young adults with cerebral palsy (CP) and their non-disabled peers; (2) to test the mediating role of financial difficulties and student status; (3) to test the moderating role of personal factors on participation difference; and (4) to test the moderating role of impairment. METHODS: A cross-sectional study was conducted in young adults [19-28 years] with CP (n = 228) and non-disabled peers (n = 2861) in France, Germany and Sweden. Participation was assessed using the Questionnaire of Young People's Participation adapted for young adults (QYPP-YA). Differences in five domains of participation were estimated using structural equation modeling with WLSMV method and bias-corrected bootstrap confidence intervals. RESULTS: Young adults with CP showed lower participation than others in all domains, with the largest difference in the "intimate relationships" domain (ß = 1.71 bcCI95[1.46; 1.95]). Student status mediated the difference in "intimate relationships", "interpersonal relationships" and "independence". Women showed greater differences than men on "independence". Impairments moderated difference in participation. The less severely impaired young adults showed no difference with their non-CP peers in "interpersonal relationships", "social life" and "independence", but made autonomous everyday decisions more often than their peers. CONCLUSIONS: Young adults with CP do not have the same opportunities to attain the participation level of non-CP people of the same age. Continuance of education could help to reduce participation difference in "interpersonal relationships" and "independence".

[Effects of High Birth Weight on the Development of Preschoolers]. / Auswirkungen eines hohen Geburtsgewichts auf die Entwicklung von Kindern im Vorschulalter.

BACKGROUND: A high birth weight above 4,000 g may lead to risks in the development of affected children. The association is less studied than the effects of very low birth weight and is the subject of this study. METHODS: The retrospective study design used data from school entry surveys in Schleswig-Holstein (Germany) collected in 2014-2017 and included 88,858 children aged 5-6 years. End points examined were language, motor skills, cognition, and behavior; use of support measures; and recommendation for special educational needs. Logistic regression models were used to estimate the association between birth weight and the outcome measures, adjusting for sociodemographic factors. RESULTS: After accounting for sociodemographic factors, high birth weight is not associated with impaired child development, whereas low birth weight emerges as a significant predictor. Across all birth weights, sociodemographic factors explain most of the variance in multivariate models of the influence of birth weight on child development. CONCLUSIONS: Very high birth weight does not justify a general recommendation for support measures. Therefore, individual developmental trajectories should be monitored. Growing up in disadvantaged circumstances may represent a significant risk and should be a reason for early intervention.

Quality of life in men with Klinefelter syndrome: a multicentre study.

Background: Klinefelter syndrome (KS) is associated with an increased risk of lower socioeconomic status and a higher risk for morbidity and mortality, which may have a significant impact on quality of life (QOL). The objective of this study is to investigate QOL in a large European cohort of men with KS. Design: Cross-sectional multicentre study. Methods: Two-hundred-eighteen men with KS were recruited from 14 clinical study centres in 6 European countries which participated in the European dsd-LIFE study. Male normative data from a healthy and a psychiatric reference population were used for comparison. The validated World Health Organization (WHO) QOL (WHOQOL)-BREF questionnaire was used to investigate five main domains of quality of life (WHOQOL): global, physical, psychological, environment, and social. Results: The QOL physical domain score was lower for men with KS compared to the healthy reference population (KS: 66.9; s.d. 19.4, n = 193; healthy reference population: 76.5; s.d. 16.2, n = 1324, P < 0.001) but higher compared to the psychiatric reference population (54.6; s.d. 20.6; n = 77, P < 0.001). The WHOQOL-psychological domain score was lower for men with KS compared to the healthy reference population (KS: 63.6; s.d. 17.8, n = 193; healthy reference population: 67.8; s.d. 15.6, n = 1324, P < 0.05) but higher compared to the psychiatric reference population (45.9; s.d. 26.0), n = 77, P < 0.001). The social domain score on the WHOQOL questionnaire was found to be lower in men with Klinefelter syndrome (KS) compared to the healthy reference population (KS: 60.0; s.d. 21.6, n = 193; healthy reference population: 68.2; s.d. 13.8, n = 1324, P < 0.001). However, this score was similar to that of the psychiatric reference population (61.0; s.d. 17.0, n = 77, P = 0.5). The WHO environment domain score of men with KS (70.0; s.d. 15.0, n = 193) was similar to the healthy reference population (70.5; s.d. 20.7, n = 1324) but higher compared to the psychiatric reference population (61.9; s.d. 20.8, n = 77, P = 0.002). Experienced discrimination, less social activities, and the presence of chronic health problems were associated with significantly decreased QOL in men with KS. Conclusion: Overall QOL in European men with KS is significantly worse compared to a healthy European reference population. Especially, the presence of discrimination, less social activities, and chronic health problems is associated with lower physical, psychological, and social QOL. Further studies are necessary to investigate if a multidisciplinary approach may help to provide adequate counselling and psychosocial support to improve QOL.

[The biopsychosocial understanding of health disorders and impairments in children with a focus on developmental neurological relationships]. / Das biopsychosoziale Verständnis von Gesundheitsstörungen und Beeinträchtigungen bei Kindern mit Fokus auf entwicklungsneurologische Zusammenhänge.

The distinction between mental (spiritual and psychological) and physical health disorders is particularly difficult due to the special features of neurobiological development in children and adolescents. In this review article, the basics of developmental neurology are briefly described. On the basis of some congenital or early acquired neurological diseases, it is then shown to what extent mental processes can be impaired in interactions with the social context. Taking these aspects into account plays an important role in child and family-oriented counseling and support. The common occurrence of physical, mental, and psychological development disorders, which is also very variable between individuals and fluctuates over the course of a person's life, requires good interdisciplinary cooperation between conservative and surgical child and adolescent medicine and child and adolescent psychiatry.

[Transition-oriented patient education program for adolescents and young adults with ADHD]. / Transitionsorientierte Patientenschulung bei Adoleszenten und jungen Erwachsenen mit ADHS.

Transition-oriented patient education program for adolescents and young adults with ADHD Abstract. Background: The transition from child- to adult-centered treatment includes numerous challenges in the treatment of chronic disorders. This process can be further complicated by disease-specific characteristics of attention-deficit/hyperactivity disorders (ADHD). This secondary analysis evaluated a transition workshop in individuals with ADHD. Methods: In total, 56 adolescents and young adults with ADHD (age M = 17.3 years, SD = 1.1; 17.9 % female) and their parents were quasi-randomly assigned to a control group (CG, n = 28) or an intervention group (IG, n = 28). The CG received regular medical care, whereas the IG additionally participated in a one-and-a-half-day transition workshop (ModuS-T). Before and 4 weeks after the intervention, transition competence was assessed with the Transition Competence Scale (TKS), patient activation with the Patient Activation Measure 13 for Adolescents (PAM® 13), and satisfaction with care with the Patient Satisfaction Questionnaire (ZUF-8). Results: The IG showed significantly improved transition competence (p ≤ .001) compared to the CG. There was no significant intervention effect in terms of patient activation (p = .194). Overall, the IG was highly satisfied with the workshop. Discussion: To date, transition workshops have been evaluated predominantly in individuals with chronic somatic disorders. This secondary analysis indicates that a generic workshop is also associated with improved transition competence and high satisfaction in individuals with chronic mental disorders. The integration of such approaches into routine care needs to be discussed.

["I Didn't Think I would be Put under Pressure Like That." Supporting and Inhibiting Factors on Access Paths to Early Prevention Programs from Parents' View]. / "So unter Druck gesetzt zu werden, das hab' ich nicht gedacht." Fördernde und hemmende Einflüsse auf Zugangswege zu den Frühen Hilfen aus Sicht der Eltern.

BACKGROUND: The German early prevention programs called "Frühe Hilfen" are open to all families with children up to 3 years old and focus on families experiencing psychosocial stress and in need of support. Families with strong psychosocial risk factors are less likely to gain access to support programs than families with lower psychosocial risk factors. OBJECTIVE: This study identifies enabling and inhibiting factors for access to early prevention programs. MATERIALS AND METHODS: Qualitative partially structured interviews were carried out with parents (N=17) from families (N=13) who had taken part in psychosocial counseling after a professional had identified support needs and recommended an early prevention program. The interviews were transcribed and interpreted using content analysis according to Mayring. RESULTS AND DISCUSSION: Parents find clear and trustworthy communication during counseling sessions to be conducive to finding access to support programs. A barrier emerges when parents feel put under pressure or do not feel a need for support. Counseling should be offered at different points in time (e. g. pregnancy, puerperium). CONCLUSION: Open, trusting, and judgement-free communication in combination with participative decisionmaking is a promising approach when it comes to parent counseling.

The impact of Klinefelter syndrome on socioeconomic status: a multicenter study.

Klinefelter syndrome (KS) is associated with an increased risk of neuropsychological morbidity, such as learning disabilities, which may have a significant impact on socioeconomic status (SES). The objective of this study was to investigate the SES in men with KS and to associate this outcome with social participation, age at diagnosis, testosterone therapy and physical and mental health status. Men with KS were recruited in 14 clinical study centers in six European countries which participated in the European dsd-LIFE study. Two hundred five men with KS were eligible for inclusion. Male normative data from the European Social Surveys (ESS) were used for comparison. Data related to education, occupation, satisfaction with income and householding were collected. Compared to the ESS reference population, fewer men with KS achieved a high level of education (13% vs 25%, P < 0.001). There was a significant difference in having a paid job (55% vs 66%, P < 0.001), and the percentage of absence by sickness or disability was higher among men with KS (10% vs 3%, P < 0.001). Furthermore, satisfaction with current household's income was lower (32% vs 42%, P < 0.01). Lower scores for subjective general health were associated with lower scores for these outcomes. Men with KS achieve on average lower levels of education, occupation and report less satisfaction with income compared to the ESS reference population. The presence of health problems and lower scores of subjective general health was related to lower levels of occupation and lower satisfaction with income in men with KS.

Two-Year Follow-Up of a Transition-Specific Education Program for Young People With Chronic Conditions.

PURPOSE: The transition to adulthood is a challenging process for young people (YP) with chronic conditions. Patient education can help to prepare them for autonomous disease management and transfer to adult healthcare services. In a previous controlled study, participation in the youth-specific group education program ModuS-T immediately improved transition-specific knowledge, competencies, and patient activation of YP with various chronic conditions. To evaluate whether these effects are long lasting, we questioned YP from the intervention group (IG) and the control group (CG; no program) 2 years after participation. METHODS: Standardized questionnaires were used to assess YP's transition-specific knowledge, competencies, patient activation, health-related quality of life, and medical care status at baseline and 2 years later. Repeated measurement covariance analyses were used for statistical testing. RESULTS: A total of 181 YP (109 IG, 72 CG) completed the questionnaires (60% of previous interviewees; mean age 20 years, 46% female). Compared to the CG, the IG reported significant improvements in transition-specific knowledge, competencies, and patient activation (p < .001) and lower parents' responsibility for their condition (p = .004). The proportion of YP who had already transferred to adult health care was nearly the same (54% IG/56% CG), but the CG was more likely to lose access to medical care (6% IG/14% CG). There were no effects on health-related quality of life. DISCUSSION: The 1.5-day ModuS-T program has long-lasting positive effects on knowledge and competencies relevant for the transition of YP. Transition-specific patient education programs close an important care gap and should be integrated into standard care.

Quality of life and mental health in emerging adults with cerebral palsy compared to the general population.

BACKGROUND: While evidence concerning Quality of Life (QoL) in youth with cerebral palsy (CP) in comparison to the general population has been accumulating, there is a lack of studies exploring differences on a wider range of positive and negative mental health outcomes in emerging adults. METHODS: This binational case control study is part of the SPARCLE cohort study on QoL and participation of youth with CP. QoL (WHOQOL-BREF), depression (PHQ-9), anxiety (GAD-7) and self-efficacy (GSE) were assessed in 198 emerging adults with CP and 593 emerging adults from the general population, matched for country of residence, age and gender. ANCOVAs with impairment and pain as covariates were run. RESULTS: Similar levels of QoL were found in both samples, except for the environmental domain, with better QoL for emerging adults with CP compared to the general population. There were significant descriptive differences regarding depression with worse levels in the CP sample, however, also worse levels of self-efficacy. Pain as a covariate had a significant negative impact on all measures, leading to poorer self-efficacy while worsening depression and anxiety; impairment had a significant worsening impact on physical QoL and self-efficacy only. CONCLUSION: Similar expressions of mental health outcomes in emerging adults with CP and the general population indicate the high adaptive capability of emerging adults with CP.

Quality of Life in Young Adults With Cerebral Palsy: A Longitudinal Analysis of the SPARCLE Study.

Introduction: While most people with cerebral palsy (CP) will have a life expectancy similar to that of the general population, international research has primarily focused on childhood and adolescence; and knowledge about the quality of life (QoL) of young adults with CP, its trajectories, and associated factors remains scarce. Methods: This longitudinal study included young adults with CP living in five European regions and who had previously participated in the SPARCLE cohort as children and/or adolescents. Their QoL in the psychological well-being and social relationships domains was estimated using age-appropriate validated instruments (KIDSCREEN-52 in childhood/adolescence and WHOQOL-Bref in young adulthood). We used generalized linear mixed-effect models with random intercept to estimate long-term trajectories of QoL in both domains and to investigate whether severity of impairment, pain, and seizure influenced these trajectories. We sought to identify potentially different trajectories of QoL from childhood to adulthood using a shape-based clustering method. Results: In total, 164 young adults with CP aged 22-27 years participated in the study. Psychological well-being linearly decreased by 0.78 points (scale 0-100) per year (95% confidence interval (CI) -0.99 to -0.56) from childhood to young adulthood, whereas QoL in the social relationships domain increased (ß coefficient 1.24, 95% CI 0.92-1.55). Severity of impairment was associated with reduced QoL in all life periods of the study (childhood, adolescence, and young adulthood): motor impairment with social relationships, and more nuancedly intellectual disability with psychological well-being and social relationships. At all periods, frequent pain significantly reduced psychological well-being, and seizures were associated with lower QoL in the social relationships domain. In both domains, we identified a group of individuals with CP who presented a reverse trajectory compared with the general QoL trajectory. Conclusion: Identification of QoL trajectories and their associated factors yields improved knowledge about the experience of individuals with CP until young adulthood. Further studies are needed to better understand the determinants that have the greatest influence on the different shapes of long-term trajectories of QoL.

A Short Form of the Child/Youth Health Care Questionnaire on Satisfaction, Utilization, and Needs in Children and Adolescents with a Chronic Condition (CHC-SUN-SF/YHC-SUN-SF).

We decided to develop a short-form of the CHC-SUN/YHC-SUN, a questionnaire aiming at assessing health care satisfaction of children and adolescents with chronic health conditions. Data analysis was based on samples from three different studies. Item selection involved statistical analysis and expert consensus. For independent validation purposes, we calculated descriptive statistics on single-item and composite-scale levels and applied classic test theory, confirmatory factor analyses, and correlation analysis to investigate the psychometric properties of the final short-form by different types of reliability and validity. Internal consistency (Cronbach's Alpha) reached values of a = 0.89 (self-report) and a = 0.92 (parents report), split-half reliability values reached 0.85 (self-report) and 0.91 (parents report). Confirmatory factor analysis indicated no sufficient fit for the single factor solution, whereas the solution with three factors and one higher order factor indicated the best overall fit amongst three competing models. Validity of the short-form measure can be assumed, e.g., as indicated by its association with a single-item measure on general health care satisfaction. The short-form measures of the CHC-SUN for parents (CHC-SUN-SF) and the YHC-SUN self-report version for adolescents (YHC-SUN-SF) feature excellent psychometric performances, provide economical assessments, and are easy-to-administer questionnaires. They should be used whenever brief measures are needed for economic reasons.

Determinants of participation and quality of life of young adults with cerebral palsy: longitudinal approach and comparison with the general population - SPARCLE 3 study protocol.

BACKGROUND: Effective inclusion in society for young people with disabilities is increasingly seen as generating opportunities for self-development, and improving well-being. However, significant barriers remain in the vast majority of activities meaningful for young adults. Research argues that various personal (disabilities, health) and environmental (access to the resources needed, accessible environment, discrimination, lack of personal economic independence) factors contribute to limited participation. However, previous studies conducted in young people with cerebral palsy (CP) mainly investigated the transition period to adulthood, and did not fully consider the whole range of impairment severity profiles or environmental barriers. In this study, we will use the follow-up of the SPARCLE cohort and a comparison group from the general population (1) to investigate the impact of the environment on participation and quality of life of young adults with CP, (2) to determine predictors of a successful young adulthood in educational, professional, health and social fields, (3) to compare quality of life and frequency of participation in social, work and recreational activities with the general population, (4) to document on participation and quality of life in those with severe disabilities. METHODS: The SPARCLE3 study has a combined longitudinal and cross-sectional design. Young adults with CP aged 22 to 27 years in 6 European regions previously enrolled in the SPARCLE cohort or newly recruited will be invited to self-complete a comprehensive set of questionnaires exploring participation (daily life and discretionary activities), health-related quality of life, body function, personal factors (health, personal resources), and contextual factors (availability of needed environmental items, family environment, services provision) during home visits supervised by trained researchers. Proxy-reports or adapted questionnaires will be used for those with the most severe impairments. The recruitment of a large group from the general population (online survey) will enable to identify life areas where the discrepancies between young people with CP and their able-bodied peers are the most significant. DISCUSSION: This study will help identify to what extent disabilities and barriers in environment negatively affect participation and quality of life, and how previous valued experiences during childhood or adolescence might modulate these effects.

["I Think it is Important in Many Aspects in Life" - Young Adults' Perception of Participation]. / "Ich finde, das ist halt in vielen Punkten im Leben wichtig" ­ Subjektive Vorstellungen junger Erwachsener von Teilhabe.

PURPOSE: This study explores young adults' lay perception of participation. It focuses on the definition of the concept and the life areas in which the target group views it as important to take part in. METHODS: In guided group interviews (N=6), 20 young adults with and without disabilities were surveyed about their perception of participation. To also include people with more severe disabilities, proxy interviews (N=3) were conducted. The data of the interview were analyzed via qualitative content analysis based on Mayring. RESULTS: Participation is understood as being multifaceted and individually different. Six aspects of participation definition could be detected: togetherness; social inclusion/participation in society; open-mindedness/interest; equal opportunities/equal treatment; freedom of choice/autonomy; to have a voice. Besides that, seven areas of participation were mentioned as important for young adults: social environment; work/education; leisure; media; mobility; politics/finances; self-administration. CONCLUSION: Within young adults' complex perception of participation, social aspects are particularly important, besides equal rights and autonomy. Participation in sexuality and new media is given a crucial value, which is why these topics should be given more attention to. Life areas in which participation is described as relevant can vary depending on the interests of the individual. This demonstrates the subjective character of the concept, and the importance to consider the perception and preference of a person or a group.

Early Genital Surgery in Disorders/Differences of Sex Development: Patients' Perspectives.

Controversy continues over a proposed moratorium on elective genital surgery in childhood for disorders/differences of sex development (DSD). Empirical evidence on patient preference is needed to inform decision-making. We conducted a multicentre survey by cross-sectional questionnaire in 14 specialized clinics in six European countries. The sample comprised 459 individuals (≥ 16 years) with a DSD diagnosis, including individuals with congenital adrenal hyperplasia (CAH) (n = 192), XY DSD with prenatal androgen effect (A) (n = 150), and without (nA) (n = 117). Main outcome measures were level of agreement with given statements regarding genital surgery, including clitoris reduction, vaginoplasty, and hypospadias repair. A total of 66% of individuals with CAH and 60% of those with XY DSD-A thought that infancy or childhood were the appropriate age for genital surgery. Females with XY DSD were divided on this issue and tended to prefer vaginoplasty at a later age (XY DSD-A 39%, XY DSD-nA 32%). A total of 47% of males preferred early hypospadias surgery. Only 12% (CAH), 11% (XY DSD-A), and 21% (XY DSD-nA) thought they would have been better off without any surgery in childhood or adolescence. Individuals who had early genital surgery were more likely to approve of it. Outcome data failed to support a general moratorium on early elective genital surgery. Participant perspectives varied considerably by diagnostic category, gender, history of surgery, and contact with support groups. Case-by-case decision-making is better suited to grasping the ethical complexity of the issues at stake.Trial registration: German Clinical Trials Register DRKS00006072.

Assessing the health-related management of people with differences of sex development.

PURPOSE: Health care requirements and perception of people with differences of sex development (DSD) have changed enormously since the "Chicago Consensus Conference" in 2005. Therefore, new standards of care and evaluation of care have to be developed. METHODS: We summarize the social and legal approach to care for DSD during the last two decades and report the main results of European research activities. RESULTS: The last two decades were accompanied by legal and societal discussion regarding how to deal with a nonbinary concept of sex. This leads to the necessity to assess health care requirements for individuals with DSD in an objective manner. We briefly review the results of the recently funded European research projects dealing with health-related issues in DSD like EU COST Action DSD, I-DSD, and dsd-LIFE, and address the compilation of quality indicators that will be needed to benchmark health care provision and health care-related outcomes. CONCLUSIONS: The benchmarking process has to be implemented among health care providers for individuals with DSD within the European Reference Networks for Rare Conditions.

The influence of psychosocial and sexual wellbeing on quality of life in women with differences of sexual development.

Background: Previous research indicating that women with differences of sexual development (DSD), namely women with Turner syndrome (TS), women with congenital adrenal hyperplasia (CAH), and women with XY-DSD, have an impaired psychosocial and sexual well-being and quality of life (QOL), was often limited by small samples and inadequate control groups (CGs). Only few studies analysed which psychosocial and sexual factors influence QOL in women with DSD and no study so far has examined whether the DSD-condition itself and the diagnostic group to which they belong moderate this influence. Methods: We compared 301 women with TS, 221 women with CAH and 142 women with XY-DSD with 603 non-DSD women regarding depression, anxiety, self-esteem, attention deficit hyperactivity disorder, autism, social participation, body acceptance, relationship status, sexual satisfaction and QOL. Furthermore, we investigated the influence of psychosocial and sexual well-being on QOL within and between diagnostic groups and examined whether the DSD-condition moderates the influence of psychosocial and sexual well-being on QOL. Results: Women with DSD reported average psychosocial well-being and QOL; only women with CAH reported an impaired physical QOL. However, women with DSD were less satisfied with their body and had less often a partner than women in the CG. Women with CAH and XY-DSD were less satisfied with their sex life compared to women in the CG. Across groups, better health and lower depression scores predicted better QOL, whereas higher self-esteem especially predicted better QOL in women with DSD. The presence of DSD moderated the influence of psychosocial and sexual well-being on QOL, however, the specific diagnosis group mainly moderated the influence on physical QOL. Conclusion: We have learned that body and sexual satisfaction need further attention in women with DSD. To optimize their QOL, psychosocial well-being should be taken in account. The improvement of self-esteem seems particularly relevant for women with DSD, as this helps coping with having a variant of sexual development.