Distress Trajectories for Parents of Children With DSD: A Growth Mixture Model.

OBJECTIVE: This study identifies trajectories of parent depressive symptoms after having a child born with genital atypia due to a disorder/difference of sex development (DSD) or congenital adrenal hyperplasia (CAH) and across the first year postgenitoplasty (for parents who opted for surgery) or postbaseline (for parents who elected against surgery for their child). Hypotheses for four trajectory classes were guided by parent distress patterns previously identified among other medical conditions. METHODS: Participants included 70 mothers and 50 fathers of 71 children diagnosed with a DSD or CAH with reported moderate to high genital atypia. Parents were recruited from 11 US DSD specialty clinics within 2 years of the child's birth and prior to genitoplasty. A growth mixture model (GMM) was conducted to identify classes of parent depressive symptoms over time. RESULTS: The best fitting model was a five-class linear GMM with freely estimated intercept variance. The classes identified were termed "Resilient," "Recovery," "Chronic," "Escalating," and "Elevated Partial Recovery." Four classes have previously been identified for other pediatric illnesses; however, a fifth class was also identified. The majority of parents were classified in the "Resilient" class (67.6%). CONCLUSIONS: This study provides new knowledge about the trajectories of depressive symptoms for parents of children with DSD. Future studies are needed to identify developmental, medical, or familial predictors of these trajectories.

Illness Uncertainty Longitudinally Predicts Distress Among Caregivers of Children Born With DSD.

OBJECTIVE: A subset of parents of children with disorders/differences of sex development (DSD) including ambiguous genitalia experience clinically elevated levels of anxious and depressive symptoms. Research indicates that uncertainty about their child's DSD is associated with parent psychosocial distress; however, previous studies have been cross-sectional or correlational in nature. The current study is the first to examine the longitudinal trajectory of the relationship between caregiver-perceived uncertainty about their child's DSD and caregiver anxious and depressive symptoms across the first 12 months following genital surgery in young children, or if surgery was not performed, the first 12 months following study entry. METHODS: One hundred and thirteen caregivers (Mage = 32.12; 57.5% mothers; 72.6% Caucasian) of children (N = 70; Mage = 9.81 months; 65.7% female) with DSD were recruited from 12 DSD specialty clinics in the United States. Caregivers completed psychosocial measures at baseline, 6 and 12 months following genitoplasty, or study entry if parents elected not to have surgery for their child. RESULTS: Caregiver illness uncertainty and both anxious and depressive symptoms were highest at baseline and decreased over time (ps < .05). Caregiver illness uncertainty predicted symptoms of anxious and depressive symptoms across all time points (ps < .05). CONCLUSIONS: Caregivers' perceptions of uncertainty about their child's DSD are highest soon after diagnosis, and uncertainty continues to predict both anxious and depressive symptoms across time. Thus, the initial diagnostic period is a critical time for psychological assessment and intervention, with parent illness uncertainty being an important clinical target.

Predictors of Psychosocial Distress in Parents of Young Children with Disorders of Sex Development.

PURPOSE: We evaluated demographic, financial and support predictors of distress for parents of young children with disorders of sex development including atypical genital development, and characterized early parental experiences. This work extends our previous findings to identify those parents at risk for distress. MATERIALS AND METHODS: Participants included mothers (76) and fathers (63) of a child (78) diagnosed with disorders of sex development characterized by moderate to severe genital atypia. Parents completed a demographic questionnaire, measures of anxious and depressive symptoms, quality of life, illness uncertainty and posttraumatic stress symptoms, and rated their satisfaction with the appearance of their child's genitalia. RESULTS: Depressive and posttraumatic stress symptoms of caregivers were comparable to standardized norms while levels of anxious symptoms were below norms. A subset of parents reported clinically elevated symptoms. Overall 26% of parents reported anxious symptoms, 24% reported depressive symptoms and 17% reported posttraumatic stress symptoms. Levels of illness uncertainty were lower than those of parents of children with other chronic illnesses. Differences by parent sex emerged, with mothers reporting greater distress. Lower income, increased medical care and travel expenses, and having no other children were related to increased psychosocial distress. CONCLUSIONS: Early psychosocial screening is recommended for parents of children with disorders of sex development. Clinicians should be aware that financial burden and lack of previous parenting experience are risk factors for distress.

Uncertainty and Posttraumatic Stress: Differences Between Mothers and Fathers of Infants with Disorders of Sex Development.

Parents of children with disorders of sex development (DSD) report significant psychological distress, including posttraumatic stress symptoms (PTSS), with mothers consistently reporting higher rates of psychological distress than fathers. However, psychological factors contributing to PTSS in both parents are not well understood. The present study sought to fill this gap in knowledge by examining PTSS and illness uncertainty, a known predictor of psychological distress, in parents of children recently diagnosed with DSD. Participants were 52 mothers (Mage = 32.55 years, SD = 5.08) and 41 fathers (Mage = 35.53 years, SD = 6.78) of 53 infants (Mage = 9.09 months, SD = 6.19) with DSD and associated atypical genital development. Participants were recruited as part of a larger, multisite study assessing parents' psychosocial response to their child's diagnosis of DSD. Parents completed measures of illness uncertainty and PTSS. Mothers reported significantly greater levels of PTSS, but not illness uncertainty, than fathers, and were more likely than fathers to report clinical levels of PTSS (21.2% compared to 7.3%). Hierarchical regression revealed that parent sex, undiagnosed or unclassified DSD status, and illness uncertainty were each associated with PTSS. The overall model accounted for 23.5% of the variance associated with PTSS. Interventions targeting illness uncertainty may be beneficial for parents of children with newly diagnosed DSD.

Psychological Adjustment of Parents of Children Born with Atypical Genitalia 1 Year after Genitoplasty.

PURPOSE: We examined the psychological adjustment of parents of children born with moderate to severe genital atypia 12 months after their child underwent genitoplasty. MATERIALS AND METHODS: Parents were recruited longitudinally from a multicenter collaboration of 10 pediatric hospitals with specialty care for children with disorders/differences of sex development and/or congenital adrenal hyperplasia. Parents completed measures of depressive and anxious symptoms, illness uncertainty, quality of life, posttraumatic stress and decisional regret. RESULTS: Compared to levels of distress at baseline (before genitoplasty) and 6 months after genitoplasty, data from 25 mothers and 20 fathers indicated significant improvements in all psychological distress variables. However, a subset of parents continued endorsing clinically relevant distress. Some level of decisional regret was endorsed by 28% of parents, although the specific decision that caused regret was not specified. CONCLUSIONS: Overall the majority of parents were coping well 1 year after their child underwent genitoplasty. Level of decisional regret was related to having a bachelor's level of education, increased levels of illness uncertainty preoperatively and persistent illness uncertainty at 12 months after genitoplasty but was unrelated to postoperative complications.

Recommendations for the Establishment of Disorders/Differences of Sex Development Interdisciplinary Care Clinics for Youth.

PURPOSE: Provide recommendations for the development of an interdisciplinary care (IDC) clinic for the treatment of youth with disorders/differences of sex development (DSD). DSD consist of a group of complex congenital medical disorders in which the development of chromosomal, gonadal, or anatomical sex is atypical. Youth with DSD require care from multiple specialized healthcare disciplines, including several medical specialties, surgery, nursing, and mental health. METHOD: Recommendations are based on an interdisciplinary care clinic model that allows for a team of relevant professionals who share knowledge, ideas, and responsibility of care. The framework established in this article is based largely on experiences at an established DSD clinic, as well as observations of multiple clinics across the United States. RESULTS: Preliminary outcome data on clinic adherence to treatment protocol under an IDC model are provided. CONCLUSIONS: To meet the diverse healthcare needs of youth with DSD, comprehensive care clinics are recommended; however, few such clinics exist in the United States. Establishing new comprehensive DSD clinics can be challenging due to the highly unique treatment of DSD, but the current paper expands the literature available to guide clinic development in the United States.

Evidence of Need to Use Self-Report Measures of Psychosocial Functioning in Older Children and Adolescents with Voiding Dysfunction.

PURPOSE: Previous reports have revealed significantly higher rates of psychosocial difficulties in children and adolescents with voiding dysfunction compared to their healthy peers. However, these findings are based solely on parental reporting and do not include self-reporting of psychosocial problems in older pediatric patients. MATERIALS AND METHODS: We collected data from 200 consecutive patients 11 to 16 years old during outpatient clinic visits. Patients completed the Pediatric Symptom Checklist-Youth Report, parents completed the parental report of the same measure, and patients and parents collaboratively completed the Dysfunctional Voiding Scoring System. RESULTS: Of the patients 25.5% met the cutoff score for clinically significant levels of psychosocial difficulties. However, only a fourth of those patients met the cutoff on the parent and self-report measures. Additionally patient self-reports of internalizing and externalizing problems were significantly related to severity of voiding dysfunction. CONCLUSIONS: Screening for psychosocial problems in older children and adolescents with voiding dysfunction should include reports from the parent and the child. In our sample 37.3% of patients needing a mental health followup would have been missed if only 1 version of the measure had been administered.

Postoperative urinary retention in men is common after carotid endarterectomy and is associated with advanced age and prior urinary tract infection.

OBJECTIVE: This study was undertaken to analyze the occurrence of postoperative urinary retention (POUR) after carotid endarterectomy (CEA) and determine whether there are any associated modifiable risk factors. CEA was chosen to minimize the confounding effects of known risk factors for POUR, including immobilization, regional and severe pain, and neuroaxial anesthesia. METHODS: This was a retrospective record review of 186 male patients undergoing CEA between 2007 and 2011. Demographic, comorbidities, and operative characteristics were compared. Continuous variables are reported as median and interquartile range (IQR) and categoric variables as frequencies and proportions. Pearson χ(2) or Mann-Whitney U tests compared categoric and continuous variables, respectively. Logistic regression was used to examine univariate and multivariate odds of POUR. Multivariate analysis controlled for known predictors of urinary retention. Association with other complications was examined with the Pearson correlation coefficient. RESULTS: POUR occurred in 34 patients (18.3%). Median age and history of urinary tract infection (UTI) were significantly associated with POUR: median age was 73.0 years (IQR, 67-80 years) for those with POUR vs 69.5 years (IQR, 63-76 years) for those without (P = .047); 17.6% of patients with a history of UTI developed POUR vs 5.9% without (P = .023). These findings persisted on multivariate analysis controlling for known predictors of POUR (body mass index, history of diabetes, benign prostate hyperplasia, and prior prostate surgery): median age (odds ratio, 1.05; 95% confidence interval, 1-1.1) and history of UTI (odds ratio, 4.16; 95% confidence interval, 1.23-14.05; P = .022). The occurrence of POUR was significantly correlated with postoperative UTI: 18.8% with POUR vs 0.7% without (Pearson r = 0.369; P < .001). CONCLUSIONS: POUR requiring bladder catheterization after CEA predisposes patients to postoperative UTI and is more common in older patients and those with a history of UTI. CEA patients lack inherent risk factors for POUR and would be a useful population for prospective studies involving POUR.

National practice patterns and outcomes of pediatric nephrectomy: comparison between urology and general surgery.

PURPOSE: In adults nephrectomy is under the purview of urologists, but pediatric urologists and pediatric general surgeons perform extirpative renal surgery in children. We compared the contemporary performance and outcome of all-cause nephrectomy at pediatric hospitals as performed by pediatric urologists and pediatric general surgeons. MATERIALS AND METHODS: We queried the Pediatric Health Information System to identify patients 0 to 18 years old who were treated with nephrectomy between 2004 and 2013 by pediatric urologists and pediatric general surgeons. Data points included age, gender, severity level, mortality risk, complications and length of stay. Patients were compared by APR DRG codes 442 (kidney and urinary tract procedures for malignancy) and 443 (kidney and urinary tract procedures for nonmalignancy). RESULTS: Pediatric urologists performed more all-cause nephrectomies. While pediatric urologists were more likely to operate on patients with benign renal disease, pediatric general surgeons were more likely to operate on children with malignancy. Patients on whom pediatric general surgeons operated had a higher average severity level and were at greater risk for mortality. After controlling for differences patients without malignancy operated on by pediatric urologists had a shorter length of stay, and fewer medical and surgical complications. There was no difference in length of stay, or medical or surgical complications in patients with malignancy. CONCLUSIONS: Overall compared to pediatric general surgeons more nephrectomies are performed by pediatric urologists. Short-term outcomes, including length of stay and complication rates, appear better in this data set in patients without malignancy who undergo nephrectomy by pediatric urologists but there is no difference in outcomes when nephrectomy is performed for malignancy.

Why does adenotonsillectomy not correct enuresis in all children with sleep disordered breathing?

PURPOSE: We analyzed the outcome of nocturnal enuresis after adenotonsillectomy in children with sleep disordered breathing. We also evaluated differences in demographic, clinical, laboratory and polysomnography parameters between responders and nonresponders after adenotonsillectomy. MATERIALS AND METHODS: We prospectively evaluated children 5 to 18 years old diagnosed with sleep disordered breathing (snoring or obstructive sleep apnea syndrome) on polysomnography and monosymptomatic primary nocturnal enuresis requiring adenotonsillectomy to release upper airway obstruction. Plasma antidiuretic hormone and brain natriuretic peptide were measured preoperatively and 1 month postoperatively. RESULTS: Sleep studies were done in 46 children and 32 also underwent blood testing preoperatively and postoperatively. Mean ± SD patient age was 8.79 ± 2.41 years and the mean number of wet nights weekly was 6.39 ± 1.26. Polysomnography revealed obstructive sleep apnea syndrome in 71.7% of patients and snoring in 28.3%. After adenotonsillectomy 43.5% of patients became dry. Preoperative polysomnography findings indicated that responders, who were dry, had significantly more arousals and obstructive apnea episodes but fewer awakenings than nonresponders, who were wet. Significant increases in plasma antidiuretic hormone and significant decreases in plasma brain natriuretic peptide were seen in all children with no difference between responders and nonresponders. No difference between the groups was noted in age, gender, race, body mass index, constipation, preoperative number of wet nights weekly or type of sleep disordered breathing. CONCLUSIONS: Nocturnal enuresis resolved after adenotonsillectomy in almost half of the children with sleep disordered breathing. Those who became dry had more frequent arousal episodes caused by apnea events than those who remained wet.

Renal bladder ultrasound evaluation in monosymptomatic primary nocturnal enuresis: is it really necessary?

BACKGROUND: Published guidelines regarding radiographic imaging in the evaluation of monosymptomatic primary nocturnal enuresis (MPNE) are not followed. We aimed to evaluate the prevalence of urological abnormalities on renal/bladder ultrasound (RBUS) in children with MPNE and to compare the RBUS findings in children with and without MPNE. METHODS: Retrospective data collection in all children aged 5-17 years seen for the initial evaluation of MPNE. Control group consisted of age- and sex-matched children who had abdominal ultrasound for other than bladder-/kidney-related causes. RBUS findings were analyzed with regard to the need for intervention and/or follow-up. RESULTS: While abnormalities on RBUS were seen in 12.54% of enuretic children and in 5.38% of controls (p = 0.004), the majority of these findings were clinically insignificant. Of those with abnormalities, only 4 enuretic children (1.43%) required intervention and 8 (2.87%) needed follow-up studies. These rates were not significantly different from the controls. However, enuretic children with RBUS abnormalities appear to be more resistant to treatment than enuretic children with normal RBUS (p = 0.002). CONCLUSIONS: A small proportion of abnormalities seen on RBUS in children with MPNE require intervention and/or further evaluation. The identification of insignificant RBUS findings could lead to unnecessary additional investigations owing to parental concern. Detailed history and a voiding diary may be sufficient in the initial evaluation of children with MPNE, although RBUS may play an important role in patients who are resistant to treatment.

Factor analysis of the pediatric symptom checklist in a population of children with voiding dysfunction and/or nocturnal enuresis.

The research objective was to identify the factor structure of the pediatric symptom checklist (PSC) in children with voiding dysfunction and/or nocturnal enuresis who were seen in a pediatric urology clinic. Retrospective chart reviews were conducted for 498 consecutive patients, ages 6-16, who were seen over a 13-month period. The PSC, a 35-item measure used to screen for psychosocial difficulties, was completed by the patient's caregiver. Confirmatory factor analyses using three previous models were conducted. A four factor model comprised of internalizing, externalizing, attention problems, and chronic illness factors represented the best fit to the data. Within this population, the PSC appears to capture internalizing and externalizing problems, difficulties with attention, and possible side effects of a medical condition. This information could aid clinicians in assessing adjustment difficulties within this population and concurrently allow researchers to examine whether these specific factors are related to other relevant outcomes.

Lower health related quality of life and psychosocial difficulties in children with monosymptomatic nocturnal enuresis--is snoring a marker of severity?

PURPOSE: Sleep disordered breathing in children is linked to numerous negative psychosocial consequences, including lower health related quality of life, increased behavioral problems and impaired neuropsychological functioning. We examined whether snoring, which is the least severe form of sleep disordered breathing, or health related quality of life could account for the increased rate of psychosocial difficulty in children with monosymptomatic nocturnal enuresis. MATERIALS AND METHODS: Patients diagnosed with monosymptomatic nocturnal enuresis seen at an outpatient pediatric urology clinic completed measures of health related quality of life (Obstructive Sleep Apnea Syndrome-18-Item Questionnaire), sleep disordered breathing (Pediatric Sleep Questionnaire) and psychosocial difficulty (Pediatric Symptom Checklist). Patients were categorized into 2 groups (snoring vs no snoring) based on the Pediatric Symptom Checklist snoring subscale score. RESULTS: Included in the study were 62 males and 45 females with a mean ± SD age of 9.09 ± 2.58 years and a mean body mass index of 21.00 ± 6.93 kg/m(2) (range 13 to 49). The sample was evenly split between 56 snorers (52.3%) and 51 nonsnorers (47.7%). Compared to children with monosymptomatic nocturnal enuresis who did not snore, MANCOVA results revealed that patients with monosymptomatic nocturnal enuresis who snored had significantly more externalizing problems and total psychosocial problems, in addition to significantly more impairment in all areas of health related quality of life. CONCLUSIONS: Snoring in children with monosymptomatic nocturnal enuresis puts them at increased risk for behavioral and psychosocial problems, in addition to impaired health related quality of life. These findings support the need for future studies of the neurological links between sleep disordered breathing and monosymptomatic nocturnal enuresis.

Bladder and bowel dysfunction: evidence for multidisciplinary care.

PURPOSE: We examined the symptoms of bladder-bowel dysfunction (ie severity of voiding dysfunction and stool consistency) and psychosocial difficulties in children presenting to the pediatric urology clinic for voiding dysfunction and to the pediatric gastroenterology clinic for functional constipation. MATERIALS AND METHODS: Parents of children seen at the gastroenterology clinic were recruited during the outpatient clinic appointment, and parents of children seen at the urology clinic were randomly selected from the research database and matched to the gastroenterology sample based on age and gender of the child. All parents completed the Dysfunctional Voiding Scoring System, Bristol Stool Form Scale, Pediatric Symptom Checklist and Parenting Stress Index™-Short Form, which assessed severity of voiding dysfunction, stool consistency, level of psychosocial difficulties and level of parenting stress, respectively. RESULTS: Children seen at the urology and gastroenterology clinics did not differ significantly on any of the measures, indicating that the severity of their bladder-bowel dysfunction is similar. However, they had significantly more severe voiding dysfunction, more constipated stool and more psychosocial difficulties than historical healthy controls. Additionally, level of parenting stress was significantly correlated with patient level of psychosocial difficulties and severity of voiding dysfunction. CONCLUSIONS: Patients with bladder and bowel dysfunction represent a homogeneous group that would potentially benefit from a multidisciplinary treatment approach involving urology, gastroenterology and psychology professionals.

History of recurrent urinary tract infection is not predictive of abnormality on voiding cystourethrogram.

PURPOSE: The American Academy of Pediatrics recently recommended against routine voiding cystourethrograms (VCUGs) in children 2 to 24 months with initial febrile UTI, raising concern for delayed diagnosis and increased risk of UTI-related renal damage from vesicoureteral reflux (VUR). We assessed factors potentially associated with higher likelihood of abnormal VCUG, including UTI recurrence, which could allow for more judicious test utilization. METHODS: We retrospectively reviewed all initial VCUGs performed at Children's Hospital of Michigan between January and June, 2010. History of recurrent UTI was ascertained by evidence of two or more prior positive cultures or history of "recurrent UTI" on VCUG requisition. Outcomes assessed included rates of VUR or any urologic abnormality on VCUG. RESULTS: Two hundred and sixty-two patients met inclusion criteria. VUR was detected in 21.3 %, urologic abnormality including VUR in 27.4 %. Degree of bladder distension, department of referring physician, study indication, positive documented urine culture, and history of recurrent UTI or UTI and other abnormality were all not associated with increased likelihood of VUR or any urologic abnormality on VCUG. CONCLUSION: VUR and VCUG abnormality are no more likely when performed after recurrent UTI or for UTI plus other abnormality. This reasons against postponing VCUG until after UTI recurrence, as positive findings are no more likely in this setting.

From hypercalciuria to hypocitraturia--a shifting trend in pediatric urolithiasis?

PURPOSE: We analyzed the metabolic abnormalities in children with urolithiasis, and the relationship between diet and hypocitraturia. MATERIALS AND METHODS: A single center, retrospective analysis was conducted in all children with renal and/or ureteral calculi seen at our Multidisciplinary Stone Clinic between January 2010 and July 2011. Data at presentation were extracted from the clinical database. RESULTS: We analyzed 63 children (37 girls) with urolithiasis with a mean age of 13.43 ± 4.61 years. Of the 45 patients with 24-hour urinalysis, a metabolic risk factor was present in 68.9%, with hypocitraturia (58.1%) and hypercalciuria (48.3%) being the most common. Children with isolated hypocitraturia had lower urinary magnesium and potassium levels (1.06 ± 0.62 mg/kg and 0.53 ± 0.24 mmol/kg per day) than those with no metabolic abnormalities (1.72 ± 0.61 mg/kg and 0.68 ± 0.20 mmol/kg per day) (p = 0.015 and p = 0.132, respectively). Urinary citrate was positively correlated with urinary potassium (r = 0.50, p = 0.002) and urinary magnesium (r = 0.49, p = 0.001). Dietary analysis revealed a lower intake of magnesium and potassium in children with hypocitraturia (28.97% ± 12.25% and 15.42% ± 7.25% recommended dietary index) than in normocitraturic cases (51.06% ± 17.51% and 45.23% ± 29.49% recommended dietary index) (p = 0.042 and p = 0.056, respectively). CONCLUSIONS: The majority of children had an identifiable metabolic risk factor for urolithiasis, with hypocitraturia being the most common. This shift in metabolic trend may be a significant contributor to the increasing incidence in pediatric urolithiasis. Hypocitraturia appears to be dietary in origin, correlated with a low consumption of potassium and magnesium.

Degree of external genital malformation at birth in children with a disorder of sex development and subsequent caregiver distress.

PURPOSE: We determined whether the degree of genital malformation at birth in children with a disorder of sex development is related to subsequent caregiver distress, specifically symptoms of depression and anxiety. MATERIALS AND METHODS: A total of 66 caregivers of children with disorders of sex development were recruited from 3 centers that specialize in disorders of sex development medicine. The caregivers completed the Beck Depression Inventory, 2nd Edition and the Beck Anxiety Inventory. The child's Prader score at birth was determined by the child's treating pediatric endocrinologist and/or pediatric urologist at each institution. RESULTS: Results from the current study revealed that for caregivers of male children, under masculinization of the child's genitals at birth was significantly related to higher levels of subsequent caregiver depression. In contrast, over masculinization of the genitals of female children at birth was unrelated to caregiver depression or anxiety. CONCLUSIONS: These findings suggest that caregivers of male children with disorders of sex development may be at increased risk for psychological distress and could benefit from family based psychosocial interventions.

The relationship of maternal and child illness uncertainty to child depressive symptomotology: a mediational model.

OBJECTIVE: To examine the relationship of parent and child ratings of illness uncertainty to depressive symptomotology in children with a chronic illness using a mediational model framework. METHOD: Mother-child dyads (N = 103 pairs) each completed measures of perceived illness uncertainty, while youth also completed a measure of depressive symptomotology. RESULTS: Maternal uncertainty was directly related to child depressive symptoms; however, this relationship was mediated by child uncertainty. CONCLUSION: It would appear that a key mechanism by which parent-related uncertainty influences child depressive symptoms is through child uncertainty, underscoring the importance of examining cognitive appraisal variables and means of transmission in parent-child interactions.

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.

Exploring Factors Associated with Decisions about Feminizing Genitoplasty in Differences of Sex Development.

STUDY OBJECTIVE: Infants with genital development considered atypical for assigned female sex may undergo feminizing genitoplasty (clitoroplasty and/or vaginoplasty) in early life. We sought to identify factors associated with parent/caregiver decisions regarding genitoplasty for their children with genital virilization. DESIGN: Longitudinal, observational study SETTING: Twelve pediatric centers in the United States with multidisciplinary differences/disorders of sex development clinics, 2015-2020 PARTICIPANTS: Children under 2 years old with genital appearance atypical for female sex of rearing and their parents/caregivers INTERVENTIONS/OUTCOME MEASURES: Data on the child's diagnosis and anatomic characteristics before surgery were extracted from the medical record. Parents/caregivers completed questionnaires on psychosocial distress, experience of uncertainty, cosmetic appearance of their child's genitalia, and demographic characteristics. Urologists rated cosmetic appearance. For 58 patients from the study cohort with genital virilization being raised as girls or gender-neutral, we compared these data across 3 groups based on the child's subsequent surgical intervention: (i) no surgery (n = 5), (ii) vaginoplasty without clitoroplasty (V-only) (n = 15), and (iii) vaginoplasty and clitoroplasty (V+C) (n = 38). RESULTS: Fathers' and urologists' ratings of genital appearance were more favorable in the no-surgery group than in the V-only and V+C groups. Clitorophallic length was greater in the V+C group compared with the V-only group, with substantial overlap between groups. Mothers' depressive and anxious symptoms were lower in the no-surgery group compared with the V-only and V+C groups. CONCLUSIONS: Surgical decisions were associated with fathers' and urologists' ratings of genital appearance, the child's anatomic characteristics, and mothers' depressive and anxious symptoms. Further research on surgical decision-making is needed to inform counseling practices.