Exploratory factor analysis of the Illness Intrusiveness Rating Scale for parents of children with atypical genital appearance due to differences of sex development (DSD).

OBJECTIVE: Illness intrusiveness refers to the subjective cognitive appraisal of a chronic health condition interfering in daily, valued activities and may be highly relevant for parents of children with atypical genital appearance due to differences of sex development (DSD). However, a measure of illness intrusiveness has not been validated for this population. The current study aimed to evaluate the factor structure of the Illness Intrusiveness Scale for Parents (IIS-P) and examine convergent validity. METHODS: Participants included 102 parents (Mage = 33.39 years, SD = 6.48; 58% mothers) of 65 children (<2 years old) diagnosed with DSD participating in a larger, longitudinal study. Parents completed the IIS-P as well as self-report measures of stigma, and anxious and depressive symptoms. An exploratory factor analysis (EFA) was conducted. RESULTS: EFA results supported a 1-factor intrusiveness solution (α = .93), as well as a 2-factor solution measuring intrusiveness on daily living (α = .92) and community connectedness (α = .85). The 1-factor solution and both factors of the 2-factor solution demonstrated significant convergent validity with stigma as well as anxious and depressive symptoms. CONCLUSIONS: Support emerged for both 1- and 2-factor solutions of the IIS-P in parents of children with DSD. The decision to evaluate illness intrusiveness as a total score or to examine the subscales of daily living and community connectedness should be tailored to the unique aims of researchers and clinicians. Future research should conduct a confirmatory factor analysis with both 1- and 2-factor models with larger, more diverse samples of caregivers.

Trajectories of illness uncertainty among parents of children with atypical genital appearance due to differences of sex development.

OBJECTIVE: The present study aimed to identify distinct trajectories of parental illness uncertainty among parents of children born with atypical genital appearance due to a difference of sex development over the first year following diagnosis. It was hypothesized that four trajectory classes would emerge, including "low stable," "high stable," "decreasing," and "increasing" classes, and that select demographic, familial, and medical factors would predict these classes. METHODS: Participants included 56 mothers and 43 fathers of 57 children born with moderate to severe genital atypia. Participants were recruited from eleven specialty clinics across the U.S. Growth mixture modeling (GMM) approaches, controlling for parent dyad clustering, were conducted to examine classes of parental illness uncertainty ratings over time. RESULTS: A three-class GMM was identified as the best-fitting model. The three classes were interpreted as "moderate stable" (56.8%), "low stable" (33.0%), and "declining" (10.3%). Findings suggest possible diagnostic differences across trajectories. CONCLUSIONS: Findings highlight the nature of parents' perceptions of ambiguity and uncertainty about their child's diagnosis and treatment the year following their child's birth/diagnosis. Future research is needed to better understand how these trajectories might shift over the course of the child's development. Results support the development of tailored, evidence-based interventions to address coping with uncertainty among families raising a child with chronic health needs.

Body image and psychosocial outcomes in youth and young adults with differences of sex development: a multi-method study.

OBJECTIVE: Differences of sex development (DSD) can affect the physical health, appearance, and psychosocial functioning of affected individuals, but little is known about how subjective appearance perceptions (body image) impact psychosocial outcomes. This study evaluated body image and its associations with psychosocial outcomes including quality of life, resilience, and psychosocial adjustment. METHODS: This cross-sectional, multi-method study assessed body image and psychosocial outcomes including quality of life, adjustment, and resilience in 97 youth and young adults with DSD (mean age = 17 ± 3.7 years; 56% assigned female in infancy) using psychometrically sound instruments. A subsample (n = 40) completed qualitative interviews. RESULTS: Quantitative results indicated that overall, participants were satisfied with their physical appearance, although less so with their primary sex characteristics. Body image dissatisfaction was associated with poorer psychosocial adjustment, quality of life, and resilience. Qualitatively, youth and young adults reported a variety of perceptions, both positive and negative, related to their body image and the impact of living with a DSD condition. Themes identified included appearance management; effects of DSD on body image; diagnostic factors and features; attitudes about diagnosis; and treatment. CONCLUSIONS: Body image is significantly associated with psychosocial outcomes in youth and young adults with DSD, with qualitative findings highlighting both positive and negative body image experiences. Results have implications for clinical care including screening for appearance concerns, normalization of appearance variations, and intervention development to better support healthy body image and psychosocial functioning in youth and young adults with DSD.

Parental Uncertainty Scale Factor Structure in Pediatric DSD With Ambiguous Genitalia.

OBJECTIVE: Illness uncertainty is a salient experience for caregivers of children with disorders/differences of sex development (DSD) presenting with ambiguous genitalia; however, no validated measure of illness uncertainty exists for this unique population. Thus, the current study aimed to preliminarily identify the factor structure of the Parental Perception of Uncertainty Scale (PPUS) in caregivers of children with DSD presenting with ambiguous genitalia and examine the convergent validity of the PPUS. METHODS: Participants included 115 caregivers (Mage = 32.12 years, SD = 6.54; 57% mothers) of children (<2-year-olds) diagnosed with DSD participating in a larger, longitudinal study. Caregivers completed the PPUS as well as self-report measures of anxious, depressive, and posttraumatic stress symptoms. An exploratory factor analysis was conducted. RESULTS: Exploratory factor analysis results indicated that a 23-item 1-factor solution was the most parsimonious and theoretically sound factor structure (α = 0.92). Convergent validity analyses demonstrated further support for the use of the 23-item 1-factor solution over the original PPUS factor structure. CONCLUSION: These results demonstrate the preliminary clinical and research utility of the PPUS with caregivers of children with DSD presenting with ambiguous genitalia. The PPUS may benefit from further refinement through qualitative research and item adaptation to capture uncertainties unique to DSD presenting with ambiguous genitalia. In addition, future research should replicate the proposed factor structure using confirmatory factor analysis with a separate, larger sample of caregivers of children with DSD to confirm the factor structure.

Adverse Birth Experiences and Parent Adjustment Associated With Atypical Genital Appearance Due to Differences of Sex Development.

OBJECTIVE: Differences/disorders of sex development (DSDs) are rare, congenital conditions involving discordance between chromosomes, gonads, and phenotypic sex and are often diagnosed in infancy. A key subset of parents of children newly diagnosed with a DSD experience clinically elevated distress. The present study examines the relationship between perinatal factors (i.e., gestational age, delivery method) and trajectories of parental adjustment. METHODS: Parent participants (mothers = 37; fathers = 27) completed measures at baseline, 6- and 12-month follow-up. Multilevel linear regression controlled for clustering of the data at three levels (i.e., time point, parent, and family) and examined the relationship between perinatal factors and trajectories of depressive and anxious symptoms. Two-way interactions between perinatal factors and parent type were evaluated. RESULTS: Overall depressive and anxious symptoms decreased over time. There were significant interactions between gestational age and parent type for depressive and anxious symptoms, with younger gestational age having a stronger negative effect on mothers vs. fathers. There was a significant interaction between time and gestational age for depressive symptoms, with 36 weeks' gestational age demonstrating a higher overall trajectory of depressive symptoms across time compared to 38 and 40 weeks. Findings for the delivery method were not significant. CONCLUSIONS: Findings uniquely demonstrated younger gestational age was associated with increased depressive symptoms, particularly for mothers compared to fathers. Thus, a more premature birth may predispose parents of infants with DSD to distress. Psychosocial providers should contextualize early diagnosis-related discussions within stressful birth experiences when providing support.

Behavioral Health Diagnoses in Youth with Gender Dysphoria Compared with Controls: A PEDSnet Study.

OBJECTIVE: To assess the odds of a psychiatric or neurodevelopmental diagnosis among youth with a diagnosis of gender dysphoria compared with matched controls in a large electronic health record dataset from 6 pediatric health systems, PEDSnet. We hypothesized that youth with gender dysphoria would have higher odds of having psychiatric and neurodevelopmental diagnoses than controls. STUDY DESIGN: All youth with a diagnosis of gender dysphoria (n = 4173 age at last visit 16.2 ± 3.4) and at least 1 outpatient encounter were extracted from the PEDSnet database and propensity-score matched on 8 variables to controls without gender dysphoria (n = 16 648, age at last visit 16.2 ± 4.8) using multivariable logistic regression. The odds of having psychiatric and neurodevelopmental diagnoses were examined using generalized estimating equations. RESULTS: Youth with gender dysphoria had higher odds of psychiatric (OR 4.0 [95% CI 3.8, 4.3] P < .0001) and neurodevelopmental diagnoses (1.9 [1.7, 2.0], P < .0001). Youth with gender dysphoria were more likely to have a diagnosis across all psychiatric disorder subcategories, with particularly high odds of mood disorder (7.3 [6.8, 7.9], P < .0001) and anxiety (5.5 [5.1, 5.9], P < .0001). Youth with gender dysphoria had a greater odds of autism spectrum disorder (2.6, [2.2, 3.0], P < .0001). CONCLUSIONS: Youth with gender dysphoria at large pediatric health systems have greater odds of psychiatric and several neurodevelopmental diagnoses compared with youth without gender dysphoria. Further studies are needed to evaluate changes in mental health over time with access to gender affirming care.

Understanding disparities and barriers associated with pediatric transplant evaluation and time to listing: Moving toward a more comprehensive picture.

BACKGROUND: Delayed time to listing (TTL) for pediatric transplant patients is associated with increased risks of mortality and morbidity. The full range of health disparities, sociodemographic factors, and other barriers associated with delays in listing in the pediatric transplant candidate evaluation process has not been fully examined. METHODS: Retrospective chart reviews were conducted for 183 kidney, liver, and heart transplant candidates ages 0-18 who were referred for evaluation during 2012-2015. Demographic information and potential barriers (e g., social/medical factors, financial concerns) were gathered from pre-transplant evaluations and included in a comprehensive model to evaluate mechanisms that explain differences in TTL. Descriptive statistics, logistic regression models, Cox proportional hazards models, and path analysis were used for analyses. RESULTS: Candidates included 26.8% heart, 33.3% liver, and 39.9% kidney patients. The most common barrier to listing was financial (71.6%), followed by caregiver psychological or substance use (57.9%), and medical problems (49.7%). Higher age, kidney, and liver organ type (relative to the heart), and presence of social, medical, administrative/motivation, and financial barriers were all directly associated with longer TTL. Public insurance was indirectly associated with TTL through social, administrative/motivation, and financial barriers. Organ type was indirectly associated with TTL through financial barriers. CONCLUSIONS: Results suggest social problems, administrative issues, and financial issues act as mechanisms through which insurance type and liver transplant candidates face increased risk of delays in transplant listing time. There are numerous clinical implications and interventions that are warranted to reduce TTL among pediatric transplant candidates with co-occurring barriers.

Behavioral Health Diagnoses in Youth with Differences of Sex Development or Congenital Adrenal Hyperplasia Compared with Controls: A PEDSnet Study.

OBJECTIVE: To evaluate the odds of a behavioral health diagnosis among youth with differences of sex development (DSD) or congenital adrenal hyperplasia (CAH) compared with matched controls in the PEDSnet database. STUDY DESIGN: All youth with a diagnosis of DSD (n = 1216) or CAH (n = 1647) and at least 1 outpatient encounter were extracted from the PEDSnet database and propensity-score matched on 8 variables (1:4) with controls (n = 4864 and 6588, respectively) using multivariable logistic regression. The likelihood of having behavioral health diagnoses was examined using generalized estimating equations. RESULTS: Youth with DSD had higher odds of a behavioral health diagnosis (OR, 1.7; 95% CI, 1.4-2.1; P < .0001) and neurodevelopmental diagnosis (OR, 1.7; 95% CI, 1.4, 2.0; P < .0001) compared with matched controls. Youth with CAH did not have an increased odds of a behavioral health diagnosis (OR, 1.0; 95% CI, 0.9, 1.1; P = .9) compared with matched controls but did have higher odds of developmental delay (OR, 1.8; 95% CI, 1.4, 2.4; P < .0001). CONCLUSIONS: Youth with DSD diagnosis have higher odds of a behavioral health or neurodevelopmental diagnosis compared with matched controls. Youth with CAH have higher odds of developmental delay, highlighting the need for screening in both groups.

Understanding Substance use Policies and Associated Ethical Concerns: A survey of Pediatric Transplant Centers.

Psychosocial risk factors, such as substance use, have been linked to poor post-transplant outcomes for solid organ transplant patients, including poor medication adherence, increased risk for rejection, and even graft failure. Despite universal consensus that substance use is an increasing problem among youth, many pediatric transplant centers do not have policies in place to address substance use and no universal guidelines exist regarding assessment during the pre-transplant evaluation in this population. An online survey was administered via REDCap™ and directed toward medical leaders (ie, medical and surgical directors) of national heart, kidney, and liver transplant centers. Questions examined the following: perspectives on the need for a universal transplant center policy on pediatric substance use, abuse, and dependence; timing and frequency of evaluation for substance use; specific substances which would elicit respondents' concerns; and ethical concerns surrounding substance use. Data were analyzed using descriptive statistics. Data were collected from 52 respondents from 38 transplant centers, with the majority (n = 40; 77%) reporting no substance use policy in place for pediatric transplant patients. However, many endorsed concerns if a pediatric patient was found to be using specific substances. Our findings further highlight the need for a universal substance use policy across pediatric solid organ transplant centers. The results from the distributed survey will help to provide guidelines and best practices when establishing a universal policy for substance use.

Providers' perspectives on substance use among solid organ transplant candidates in a pediatric hospital.

INTRODUCTION: Substance use is prevalent among youth and often leads to impairment in multiple domains. Additionally, substance use may pose adverse health issues post-transplant. Yet, practices related to substance use among pediatric patients who require organ transplant remain inconsistent. In this study, providers were surveyed for their perspectives on substance use among solid organ transplant candidates within a pediatric hospital. METHODS: An online survey was administered to providers on the heart, kidney, and liver transplant teams at one tertiary pediatric care center located in the intermountain region of the United States (N = 50, 42% response rate). Providers answered questions about the need for a hospital-wide policy across heart, liver, and kidney transplant teams within this transplant center, timing of substance use evaluation, types of substances eliciting concerns based on organ, and recommended interventions. Data were analyzed using descriptive statistics. RESULTS: Providers felt strongly about the need for a policy to guide recommendations for substance use among transplant candidates. Providers wanted a hospital-wide substance use policy (84%) and a standardized measure for assessing substance use (98%). Respondents (98%) indicated that substance use should be assessed during the pretransplant evaluation. Respondents expressed varied concerns based on substance and organ type, and recommended interventions for patients to cease substance use prior to transplant listing. CONCLUSIONS: This study highlights the need for a clear, directive, hospital-wide policy and standardized procedure for evaluating substance use among adolescent solid organ transplant candidates nationally across pediatric transplant centers.

Implementing a Home-Based Telehealth Group Adherence Intervention with Adolescent Transplant Recipients.

Introduction: To improve access to care and engage adolescent transplant recipients for adherence-promoting interventions, innovative solutions utilizing technology are needed. Materials and Methods: This study describes the implementation of a five-session group intervention targeting medication adherence in adolescent transplant recipients through home-based telemedicine. Results: Seven videoconferencing groups were conducted with a total of 33 participants living a median of 57 miles away from their transplant medical center. The median coefficient of variation (CV) of tacrolimus (immunosuppression medication) decreased from 32.2% to 23.5% from the pregroup to postgroup phase. Analyses indicated that the group was acceptable and engaging for participants, as highlighted by one participant reporting, "I liked how me and the group members came together… it felt like a little family." Overall satisfaction with the group and with the technology was endorsed by 86%. The group intervention was generally feasible; however, there were technological difficulties reported by participants and the facilitator, particularly given the diversity of the home-based connections and equipment. Other adaptations helped improve recruitment, attendance, and participation. Discussion: Implementing a telehealth group can pose unique challenges, especially with a group of adolescent participants. Although nonsignificant, we observed a decrease in the median CV of tacrolimus, indicating that medication adherence generally improved after group intervention. Our experience facilitating these groups provides insights into strategies to optimize feasibility and the participant experience. Distance of participants from the hospital is an important consideration and provides a strong rationale for the need for telemedicine-enabled approaches. We explore and discuss the challenges to implementing a home-based group, suggest practical strategies and developmentally sensitive adjustments when working with adolescents, and propose strategies to prepare clinicians for obstacles that may present when implementing a telehealth group with youth.

Adherence in pediatric kidney transplant recipients: solutions for the system.

Non-adherence remains a significant problem among pediatric (and adult) renal transplant recipients. Non-adherence among solid organ transplant recipients results in US$15-100 million annual costs. Estimates of non-adherence range from 30 to 70% among pediatric patients. Research demonstrates that a 10% decrement in adherence is associated with 8% higher hazard of graft failure and mortality. Focus has begun to shift from patient factors that impact adherence to the contributing healthcare and systems factors. The purpose of this review is to describe problems within the systems implicated in non-adherence and potential solutions that may be related to positive adherence outcomes. Systems issues include insurance and legal regulations, provider and care team barriers to optimal care, and difficulties with transitioning to adult care. Potential solutions include recognition of how systems can work together to improve patient outcomes through improvements in insurance programs, a multi-disciplinary care team approach, evidence-based medical management, pharmacy-based applications and interventions to simplify medication regimens, improved transition protocols, and telehealth/technology-based multi-component interventions. However, there remains a significant lack of reliability in the application of these potential solutions to systems issues that impact patient adherence. Future efforts should accordingly focus on these efforts, likely by leveraging quality improvement and related principles, and on the investigation of the efficacy of these interventions to improve adherence and graft outcomes.

Medication adherence among adolescent solid-organ transplant recipients: A survey of healthcare providers.

The aim of this study was to assess healthcare provider perspectives on barriers to medication adherence and to discover recommendations for interventions among providers of pediatric solid-organ transplant patients. An anonymous online survey was administered to a multidisciplinary pool of pediatric transplant providers from February 2015 to March 2016. It consisted of 15 questions regarding transplant providers' attitudes, clinical practice, and beliefs pertaining to medication adherence among teenage solid-organ transplant recipients. Data were analyzed using descriptive statistics. Responses to open-ended questions were coded and categorized into themes. One hundred ten surveys were completed by providers specializing in pediatric heart, kidney, liver, lung, and/or intestinal transplantation. Commonly cited reasons for poor adherence were forgetting/poor planning (94%), the desire to be normal (86%), lack of support (86%), and poor parental monitoring (79%). Suggestions to improve adherence included increasing peer and family support, providing education, and incorporating technology into adherence regimens. Barriers to adherence in transplant patients are recognized by providers and are both similar to and disparate from patient and family identified barriers published in the literature. Providers recognize the importance of education, social support, and technologically driven interventions on improving outcomes in the transplant population.

Healthcare communication satisfaction and psychosocial outcomes in adolescents and young adults with differences of sex development.

OBJECTIVES: We examined: (1) healthcare communication satisfaction and psychosocial outcomes (resilience, QoL, and psychological adjustment) in adolescents and young adults (AYA) with differences of sex development (DSD), (2) differences in psychosocial outcomes between those who were highly satisfied and those who were less satisfied, and (3) group differences between adolescents (ages 12-17) and young adults (ages 18-26) regarding associations with healthcare communication and psychosocial outcomes. METHODS: AYA with DSD across four study sites reported on satisfaction with healthcare communication and psychosocial outcomes. Analyses included descriptive statistics (aim 1), independent samples t-tests (aim 2), and Pearson's correlations (aim 3). RESULTS: Participants reported high levels of satisfaction with healthcare communication. Higher healthcare communication satisfaction was associated with greater resilience (p = .01), better QoL (p = .02), and fewer internalizing problems (p = .04). For adolescents, higher healthcare communication satisfaction was associated with better psychosocial outcomes (p values ranging from.01-.04). No significant associations were found in the young adult group. CONCLUSIONS: Satisfaction with healthcare communication is related to positive psychosocial outcomes in adolescents with DSD. PRACTICE IMPLICATIONS: These data underscore the importance of optimizing communication with families, engaging AYA in early and ongoing discussions about their care, and including psychosocial providers in DSD care.

Parental experience and understanding of parent-provider discussions of treatment for infants with ureteropelvic junction obstruction.

Objective: The purpose of the current study was to understand what families identify as necessary information to guide decision-making in the treatment of their child with UPJO. Methods: We conducted semi-structured interviews with parents of children with UPJO using phenomenological methodology. Data were systematically analyzed according to principles of thematic analysis, using a team-based inductive approach. Results: 32 parents were interviewed. Findings are organized by three major themes including barriers to meaningful participation in decision making, logistical aspects of the decision, and psychosocial aspects of the decision. Conclusion: These findings suggest the need to increase parent education and understanding around medical and surgical decision-making, and the need to enhance psychosocial support for more meaningful parental engagement in the surgical decision-making process. Practice implications: The findings from the interviews highlight the importance of caregivers needing clear and accurate information in order to engage in meaningful discussions related to surgical decision-making for decisions around surgery for UPJO treatment.

Information needs and health status in adolescents and young adults with differences of sex development or sex chromosome aneuploidies.

INTRODUCTION: When and how to provide condition-related information to adolescents and young adults (AYAs) with differences of sex development or sex chromosome aneuploidies (DSDs or SCAs) is largely based on anecdotal experience and lacks informed guidance. For AYAs with a DSD or SCA, having accurate information is critical for attaining optimal adjustment and well-being, participating in decision making related to treatment options, and transitioning successfully to adult health care, yet prior studies have focused exclusively on parental perspectives and not on the views of adolescents themselves. OBJECTIVE: The objective of this study was to describe unmet information needs in AYAs with a DSD or SCA and examine associations with perceived global health. METHODS: Participants were recruited from specialty clinics at Children's Hospital of Philadelphia (n = 20) and Children's Hospital Colorado (n = 60). AYAs ages 12-21 years with a DSD or SCA and a parent completed a survey assessing perceived information needs across 20 topics, importance of those topics, and global health using the PROMIS Pediatric Global Health questionnaire (PGH-7). RESULTS: AYAs had diagnoses of Klinefelter syndrome (41%), Turner syndrome (25%), and DSD (26%) and were 16.7 years (SD = 2.56) and 44% female. Parent participants were primarily mothers (81%). AYAs perceived that 48.09% of their information needs were unmet (SD = 25.18, range: 0-100). Parents perceived that 55.31% of AYAs' information needs were unmet (SD = 27.46 range: 5-100). AYAs and parents across conditions reported unmet needs related to information about transition to adult health care, financial support for medical care, and how the condition might affect the AYA's health in the future. While AYA-reported PGH-7 scores were not associated with percentage of AYA unmet information needs, parent-reported PGH-7 scores were (r = -.46, p < .001), such that lower parent-reported global health was associated with higher percentage of AYA unmet information needs. DISCUSSION/CONCLUSION: On average, parents and AYAs perceived that half of AYAs' information needs were unmet, and a higher percentage of AYA unmet information needs was associated with lower perceived global health. The frequency of unmet needs in this sample of AYAs reflects an opportunity for improvement in clinical care. Future research is needed to understand how education to children and AYAs unfolds as they mature and to develop strategies to address the information needs of AYAs with a DSD or SCA, promote well-being, and facilitate AYA engagement in their own health care.

Unexpected challenges faced by caregivers of children with neurogenic bladder: A qualitative study.

BACKGROUND: Management of the neurogenic bladder is variable, complex, and often requires a demanding bladder care regimen which may present caregiver burdens that are unique among chronic disease. While research into patient quality of life is increasing, parallel study of the caregiver experience is scant. Existing research primarily comprises survey data using validated instruments originally developed for non-urologic conditions, such as dementia. These surveys may detect high caregiver burden and decreased quality of life amongst caregivers but are limited in their ability to understand the underlying causes. OBJECTIVE: To characterize the experience of those caring for children with neurogenic bladders, with a focus on unexpected burdens and challenges. METHODS: In light of limited existing research, a qualitative research methodology was selected to explore the caregiver experience. Semi-structured phone interviews were conducted with primary caregivers of children with neurogenic bladder, all of whom were patients in the pediatric urology department of a single tertiary pediatric referral center. Purposive sampling was used to ensure diverse representation. Interviews were recorded, transcribed, and professionally translated if needed. Transcripts were analyzed using a team-based inductive grounded-theory approach, facilitated by ATLAS. ti software. Member-checking focus groups were held to validate the results. RESULTS: Twenty-five caregivers were interviewed (20 in English, 5 in Spanish), at which point thematic saturation was reached. Three primary themes emerged surrounding the topic of unexpected challenges: 1. High caregiver burden, 2. Challenges with catheterization and supplies, 3. Urinary tract infections. Member-checking focus groups validated the thematic analysis and provided additional insights into mitigating factors for these challenges. A child's independence with his or her health care regimen was cited as particularly important for decreasing caregiver burden. DISCUSSION: Caregivers of children with neurogenic bladder report their role is more difficult than they anticipated it would be. Catheterization represents a particularly burdensome task, and recurrent infections are an unexpected and persistent medical challenge. Understanding unexpected challenges that caregivers face will help pediatric urologists target modifiable factors to decrease caregiver burden, address current gaps in counseling and expectation-setting, and set the stage for more complete shared decision-making. CONCLUSIONS: This study represents an initial qualitative characterization of the experience caring for a child with neurogenic bladder. This is a key first step in understanding how caregivers make decisions for their children and their families. This initial study is foundational to a larger project to create a decision aid for caregivers of children with neurogenic bladder.

Instituting Sexual Orientation and Gender Identity Training and Documentation to Increase Inclusivity at a Pediatric Health System.

In this mixed-methods quality improvement project, we implemented and evaluated sexual orientation and gender identity (SOGI) form rollout in the electronic medical record. Families in our gender diversity program completed a baseline survey in 2017 (55/328 responded) and follow-up in 2020 (180/721 responded) to evaluate the frequency of affirmed name and pronoun use in the hospital. Survey feedback informed system-wide inclusivity efforts and training. SOGI was implemented in 2020 after 1,662 providers completed an online training and 11,090 team members completed gender and sexual orientation inclusivity training. We recommend similar trainings for health systems utilizing SOGI.

Surgical experiences in adolescents and young adults with differences of sex development: A qualitative examination.

BACKGROUND: Surgical intervention in youth with differences of sex development (DSD) is a controversial topic. Historically, evidence suggests that genital surgery in DSD is associated with mixed patient satisfaction. However, less is known about surgical outcomes under therapeutic advancements in the past several decades. OBJECTIVE: The purpose of the current study is to provide an updated and empirical qualitative examination of the surgical experiences and responses to care of adolescents and young adults (AYA) with DSD in order to fill this gap in the literature and inform patient care. METHODS: Qualitative interviews were conducted with 37 AYA (ages 12-26) with DSD. Interviews were transcribed, coded, and analyzed thematically. RESULTS: Three major themes were identified: 1) knowledge related to surgery; 2) surgical and medical experience; and 3) psychosocial factors related to surgery. Results demonstrated that most AYA were not involved in the decision to pursue surgery and were observed to have varying levels of knowledge regarding their surgeries. Most participants in the current study had received a DSD-related surgery and the majority described having positive surgical experiences and few regrets. Nonetheless, AYA described both medical and psychosocial challenges related to their surgeries and recoveries and offered feedback on ways to improve the surgical process. DISCUSSION: The current study provides a qualitative examination of the surgical experiences of 37 AYA with DSD. Findings highlight the importance of regular and ongoing communication with providers to improve knowledge related to surgery during the decision-making process as well as after surgical intervention. Results underscore the benefits of multidisciplinary teams and the value of patient handouts and decision aids in assisting AYA in the decision to pursue surgical intervention. Other specific recommendations for providers include increased patient privacy during genital exams, greater emphasis on psychoeducation and pain management strategies, and the use of behavioral health services to assist with challenges and social support. CONCLUSIONS: The decision to pursue surgical intervention in youth with DSD remains a complex and controversial issue, and more information regarding patients' perspectives on surgery is needed. The current study provides novel insights into patient experiences of surgical intervention and highlights the need for psychosocial support throughout the shared decision-making process.

Decisional regret about surgical and non-surgical issues after genitoplasty among caregivers of female infants with CAH.

INTRODUCTION: Caregivers of female infants with congenital adrenal hyperplasia (CAH) often confront complex medical decision-making (e.g., early feminizing genitoplasty). OBJECTIVE: This study aimed to evaluate the relevant medical decisions and subsequent decisional regret of caregivers following their child's genitoplasty. STUDY DESIGN: Caregivers (N = 55) were recruited from multidisciplinary treatment programs for participation in a longitudinal study. Qualitative data was collected at 6-12 months following feminizing genitoplasty to evaluate caregiver-reported decision points across their child's treatment. Quantitative exploratory analysis evaluated pre-operative predictors of subsequent decisional regret. DISCUSSION: When prompted about their decision-making and potential regret, most caregivers (n = 32, 80%) reported that their daughter's genital surgery was their primary medical decision. Specific themes regarding genital surgery included the timing and type of surgery. Most caregivers reported no decisional regret (62%), with 38% reporting some level of regret. Greater pre-operative illness uncertainty predicted heightened decisional regret at follow-up, p = .001. CONCLUSION: Two-thirds of caregivers of female infants with CAH reported not regretting their decision-making. Nevertheless, over one-third of caregivers reported some level of regret, suggesting the need for improvements in shared decision-making processes. Many, but not all, families reported that this regret was related to surgical decision-making. Reducing caregiver illness uncertainty (e.g., providing clear information to families) may increase their satisfaction with decision-making. Further research is needed to determine how the evolving care practices surrounding early genitoplasty will impact families.