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.

Feasibility of Enhanced Recovery After Surgery (ERAS) implementation in Pediatric Urology: Pilot-phase outcomes of a prospective, multi-center study.

INTRODUCTION/BACKGROUND: Enhanced Recovery After Surgery (ERAS) is a fundamental shift in perioperative care that has consistently demonstrated an improved outcome for a wide variety of surgeries in adults but has only limited evidence in the pediatric population. OBJECTIVE: We aimed to assess the success with and barriers to implementation of ERAS in a prospective, multi-center study on patients undergoing complex lower urinary tract reconstruction. STUDY DESIGN: Centers were directed to implement an ERAS protocol using a multidisciplinary team and quality improvement methodologies. Providers completed pre- and post-pilot surveys. An audit committee met after enrolling the first 5 patients at each center. Pilot-phase outcomes included enrollment of ≥2 patients in the first 6 months of enrollment, completion of 90 days of follow-up, identification of barriers to implementation, and protocol adherence. RESULTS: A total of 40 patients were enrolled across 8 centers. The median age at surgery was 10.3 years (IQR 6.4-12.5). Sixty five percent had a diagnosis of myelomeningocele, and 33 % had a ventriculoperitoneal shunt. A bladder augmentation was performed in 70 %, Mitrofanoff appendicovesicostomy in 52 %, Monti ileovesicostomy in 15 %, and antegrade continence enema channel in 38 %. The most commonly perceived barriers to implementation on the pre-pilot survey were "difficulty initiating and maintaining compliance with care pathway" in 51 % followed by a "lack of time, money, or clinical resources" in 36 %. The pre-pilot study experience, implementation, and pilot-phase outcomes are provided in the Table. All primary and secondary outcomes were achieved. DISCUSSION: The findings of the present study were similar to several small comparative studies with regard to the importance of a multidisciplinary team, strong leadership, and continuous audit for successful implementation of ERAS. Similar barriers were also encountered to other studies, which primarily related to a lack of administrative support, leadership, and buy-in from other services. The limitations of the present study included a relatively small heterogeneous cohort and absence of a comparative group, which will be addressed in the larger exploratory phase of the trial. The findings may also not be generaziable due to the need for sustainable processes that were unique to each center as well as an absence of adequate volume or resources at smaller centers. CONCLUSIONS: ERAS was successfully implemented for complex lower urinary tract reconstruction across 8 centers through a multidisciplinary team, structured approach based on the local context, and focus on a continuous audit.

The real world experience of pediatric primary hyperoxaluria patients in the PEDSnet clinical research network.

The rarity of primary hyperoxaluria (PH) challenges our understanding of the disease. The purpose of our study was to describe the course of clinical care in a United States cohort of PH pediatric patients, highlighting health service utilization. We performed a retrospective cohort study of PH patients < 18 years old in the PEDSnet clinical research network from 2009 to 2021. Outcomes queried included diagnostic imaging and testing related to known organ involvement of PH, surgical and medical interventions specific to PH-related renal disease, and select PH-related hospital service utilization. Outcomes were evaluated relative to cohort entrance date (CED), defined as date of first PH-related diagnostic code. Thirty-three patients were identified: 23 with PH type 1; 4 with PH type 2; 6 with PH type 3. Median age at CED was 5.0 years (IQR 1.4, 9.3 years) with the majority being non-Hispanic white (73%) males (70%). Median follow-up between CED and most recent encounter was 5.1 years (IQR 1.2, 6.8). Nephrology and Urology were the most common specialties involved in care, with low utilization of other sub-specialties (12%-36%). Most patients (82%) had diagnostic imaging used to evaluate kidney stones; 11 (33%) had studies of extra-renal involvement. Stone surgery was performed in 15 (46%) patients. Four patients (12%) required dialysis, begun in all prior to CED; four patients required renal or renal/liver transplant.    Conclusion: In this large cohort of U.S. PH children, patients required heavy health care utilization with room for improvement in involving multi-disciplinary specialists. What is Known: • Primary hyperoxaluria (PH) is rare with significant implications on patient health. Typical involvement includes the kidneys; however, extra-renal manifestations occur. • Most large population studies describe clinical manifestations and involve registries. What is New: • We report the clinical journey, particularly related to diagnostic studies, interventions, multispecialty involvement, and hospital utilization, of a large cohort of PH pediatric patients in the PEDSnet clinical research network. • There are missed opportunities, particularly in that of specialty care, that could help in the diagnosis, treatment, and even prevention of known clinical manifestations.

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.

Infantile scrotal hemangioma with transverse testicular ectopia.

Transverse testicular ectopia is a rare structural anomaly of abnormal testicular descent. We report a case of a 3-month-old boy with hemiscrotal infantile hemangioma and contralateral transverse testicular ectopia.

Fibroepithelial polyp causing urethral obstruction: Diagnosis by cystourethrogram.

Fibroepithelial polyps of the urethra are rare benign tumors that predominantly affect males in childhood or adolescence. In this report, we present a case of a 3-year-old boy in acute urinary retention with a urethral fibroepithelial polyp manifesting as a large filling defect on voiding cystourethrogram and successfully managed endoscopically with transurethral resection.

Bladder Recurrence of Clear Cell Sarcoma of the Kidney Seven Years After Initial Presentation.

Clear cell sarcoma of the kidney (CCSK) is the second most common pediatric renal malignancy after Wilms tumor. CCSK has the potential to metastasize to distant sites and was historically known as the bone metastasizing renal tumor. We report an exceedingly rare case of a bladder recurrence of CCSK. Our patient presented with gross hematuria 7 years after initial complete response. He was found to have a large sessile bladder tumor and underwent a partial cystectomy with right pelvic lymph node dissection. Final pathology was metastatic CCSK.

Adult hypospadias: urethral and penile reconstruction.

PURPOSE OF REVIEW: This article provides a subjective, concise review of contemporary advances in reconstructive urology as it pertains to adult hypospadias repair. Herein, we highlight the most important and interesting articles among the many published within the past 12 months. RECENT FINDINGS: The main themes in the recent literature covered herein, include management of postoperative complications, long-term follow-up, and penile cosmesis. SUMMARY: Recent literature would suggest beauty is in the eye of the beholder when it comes to cosmesis after hypospadias repair. Long-term data are reassuring regarding uroflow improvement through puberty and into adulthood for common distal shaft hypospadias repairs.

Long-term efficacy and durability of botulinum-A toxin for refractory dysfunctional voiding in children.

PURPOSE: We evaluated our long-term experience with intrasphincteric botulinum toxin A injection in children with dysfunctional voiding. MATERIALS AND METHODS: From January 2006 through July 2012 we saw 2,172 neurologically normal children due to dysfunctional voiding. Of patients who presented to these visits we retrospectively identified the charts of 12 with dysfunctional voiding (8 females) in whom urotherapy and medical management failed and who underwent botulinum toxin A injection to the external urinary sphincter. Mean patient age at surgery was 10.5 years (range 4 to 19). Average followup was 45 months (range 20 to 71). Preoperatively and postoperatively all children were evaluated with history and physical examination, voiding diary, renal and pelvic ultrasound with post-void residual volume measurement and uroflowmetry. RESULTS: Eight of the 12 children (67%) experienced significant improvement in voiding parameters. Before vs after treatment mean ± SD post-void residual urine volume was 115 ± 83 vs 57 ± 61 ml (p = 0.016) and the mean maximum flow rate was 11.8 ± 8.1 vs 20.4 ± 7.9 ml per second. Half of the cohort required a second injection an average of 15 months later. Three of the 4 patients who failed to show improvement had neuropsychiatric problems and 1 had evidence of bladder underactivity. CONCLUSIONS: Our results demonstrate reasonable efficacy and durability of intrasphincteric botulinum toxin A injection in children with refractory dysfunctional voiding. Neuropsychiatric issues appear to negatively influence the success rate. Long-term followup is vital to identify patients in whom repeat injection may be necessary.

Endopyelotomy for pediatric ureteropelvic junction obstruction: a review of our 25-year experience.

PURPOSE: We elucidate the role of endopyelotomy as a primary and secondary intervention for ureteropelvic junction obstruction in children. MATERIALS AND METHODS: We retrospectively identified 79 pediatric patients who underwent endopyelotomy for ureteropelvic junction obstruction between 1986 and 2011. Eleven patients were lost to followup and were excluded from analysis. Patient demographics, operative information, complications and success rates were reviewed for the remaining 68 patients. Treatment success was defined as the absence of symptom recurrence and improved radiographic features on ultrasound, computerized tomography, diuretic renogram or excretory urogram at most recent followup. RESULTS: Primary endopyelotomy data were analyzed in 37 patients with a median age of 11.1 years. The success rate was 65% at a median followup of 34 months (range 1.5 to 242). Treatment failure occurred in 13 patients with a median time to failure of 8 months (range 1.5 to 131). There were 8 cases of failure during 12 months of surgery. Secondary endopyelotomy data were analyzed in 31 patients with a median age of 6.5 years. The success rate was 94% at a median followup of 61 months (range 1 to 204). Treatment failure occurred in 2 patients at 1 and 6 months. Approximately two-thirds of all procedures used an antegrade approach. CONCLUSIONS: Primary endopyelotomy is significantly less successful than pyeloplasty in the treatment of ureteropelvic junction obstruction in pediatric patients. However, secondary endopyelotomy following failed pyeloplasty represents a viable alternative to redo pyeloplasty.

The efficacy of oral midazolam for decreasing anxiety in children undergoing voiding cystourethrogram: a randomized, double-blind, placebo controlled study.

PURPOSE: Voiding cystourethrogram is an invasive test that evokes anxiety. Our primary aim was to determine whether midazolam is beneficial in decreasing anxiety in children who undergo voiding cystourethrogram. Secondary aims were an examination of parent anxiety, health care professional perceptions and post-procedure behavioral outcomes in children after voiding cystourethrogram. MATERIALS AND METHODS: A total of 44 children were randomized to placebo or oral midazolam before voiding cystourethrogram in double-blind fashion. The Modified Yale Preoperative Anxiety Scale was used to evaluate child behavior before and during voiding cystourethrogram, and the Post Hospitalization Behavior Questionnaire was used to investigate any short-term and intermediate-term behavioral outcomes. The State-Trait Anxiety Inventory was used to evaluate parent personal anxiety during voiding cystourethrogram. A separate questionnaire was administered to radiology staff. Statistical analysis included the 2-sample t and Fisher exact tests. RESULTS: There was no difference in Modified Yale Preoperative Anxiety Scale scores in children randomized to midazolam or placebo. There was also no significant difference in parent anxiety. Radiology care providers identified no reliable benefit when blinded to sedation vs placebo. We did not note any post-procedural behavior issues after voiding cystourethrogram at up to 6 months of followup. CONCLUSIONS: Midazolam may not significantly help with child or parent anxiety during voiding cystourethrogram. No reliable benefit was noted according to radiology health care provider perception and there was no significant post-procedural behavior benefit. Midazolam may not provide a significant benefit in decreasing anxiety during voiding cystourethrogram.

Efficacy and safety of transdermal and oral oxybutynin in children with neurogenic detrusor overactivity.

PURPOSE: We evaluated the efficacy and safety of transdermal and oral oxybutynin in children with neurogenic detrusor overactivity. MATERIALS AND METHODS: Children with neurogenic detrusor overactivity 6 to 15 years old and previously receiving oxybutynin were assigned randomly at a 3:1 ratio to treatment with transdermal or oral oxybutynin. Initial dosages (transdermal 1.3, 2.9 or 3.9 mg daily; oral 5, 10 or 15 mg daily), based on pre-study dosages, were adjusted after 2 weeks and then maintained for 12 weeks. The primary efficacy end point was change from baseline to last observation in average urine volume collected by clean intermittent catheterization. RESULTS: A total of 57 patients were randomized to receive transdermal (41) or oral (16) oxybutynin. Safety data were available for 55 patients and efficacy data were available for 52. Mean +/- SD urine volume increased from 95 +/- 64 ml to 125 +/- 74 ml (p <0.001) with transdermal oxybutynin and from 114 +/- 75 ml to 166 +/- 92 ml (p = 0.002) with oral oxybutynin. Transdermal oxybutynin resulted in significant improvement in all measured urodynamic parameters. Similar trends and a significant increase in maximal cystometric bladder capacity were observed in the smaller oral oxybutynin group. There were 12 treatment related adverse events noted with transdermal oxybutynin (mild skin reaction) and 1 with oral oxybutynin (vasodilatation). The ratio of N-desethyloxybutynin-to-oxybutynin plasma concentrations was substantially lower with transdermal (1.4) than with oral (6.7) oxybutynin. CONCLUSIONS: Transdermal oxybutynin was a well tolerated and effective alternative to oral oxybutynin in treating neurogenic detrusor overactivity in children who previously tolerated oxybutynin.

Impact of wait time on outcome for inguinal hernia repair in infants.

BACKGROUND: Prolonged surgical wait times are a problem in many health care systems. We used data from two pediatric surgical centers, one Canadian and one American, in order to determine if increased wait times are related to rates of incarceration and adverse outcomes. METHODS: Data were collected for children under the age of 2 who presented with an inguinal hernia to either the emergency department or clinic in the two hospitals in 2002 and 2003. RESULTS: Infants in the Canadian center were older at presentation and were more likely to present to the emergency department. Wait time for hernia repair was longer in the Canadian than the American hospital (99 +/- 103 vs. 27 +/- 53 days, P < 0.001). The incidence of incarceration was higher in the Canadian hospital, and infants in the Canadian center were more likely to have episodes of recurrent incarceration. Emergency department usage was greater in the Canadian hospital both at the time of diagnosis as well as during the waiting period for surgery. DISCUSSION: Prolonged wait time for inguinal hernia repair in infants is associated with a higher rate of incarceration as well as greater usage of emergency department resources. These data are important for those surgeons working in systems with limited resources in which strategies to shorten wait times are necessary.

The incidence of disorders of sexual differentiation and chromosomal abnormalities of cryptorchidism and hypospadias stratified by meatal location.

PURPOSE: Routine karyotype analysis has been recommended for patients with cryptorchidism and hypospadias. However, it is unclear whether karyotyping should be obtained in all patients, or tailored to the severity or degree of hypospadias. Therefore, we analyzed the incidence of chromosomal abnormalities in patients with distal or proximal hypospadias and concomitant cryptorchidism. MATERIALS AND METHODS: We reviewed the records of patients with cryptorchidism and hypospadias treated at a pediatric hospital between 1994 and 2006. Data collected included karyotype analysis, gonad palpability, and meatal and testes location at time of surgery. Patients with retractile testes and congenital adrenal hyperplasia were excluded from analysis. RESULTS: We identified 44 patients with hypospadias and cryptorchidism (26 with proximal and 18 with distal hypospadias). Karyotype information was available in 25 patients (19 with proximal and 6 with distal hypospadias). None of the patients with distal hypospadias and cryptorchidism had an abnormality of a sex chromosome. In contrast, chromosomal abnormalities were present in 6 of 19 individuals (32%) with proximal hypospadias and cryptorchidism. The most common abnormality was mixed gonadal dysgenesis in 3 patients, followed by autosomal translocations in 2 and 48XY aneuploidy in 1. CONCLUSIONS: When karyotype information was stratified by meatal location with cryptorchidism we found no significant chromosomal abnormalities in distal hypospadias and cryptorchidism, whereas a third of patients with proximal hypospadias and cryptorchidism had an abnormal karyotype. Karyotype analysis appears to be important in individuals with cryptorchidism and proximal hypospadias but of little benefit in patients with distal hypospadias and palpable undescended testes.

Correlation of prenatal and postnatal ultrasound findings with the incidence of vesicoureteral reflux in children with fetal renal pelvic dilatation.

PURPOSE: Up to 1% of prenatal ultrasounds detect renal pelvic dilatation. This dilatation is associated with vesicoureteral reflux but its clinical significance and the necessity for vesicoureteral reflux detection have been questioned. We report an evaluation of fetal renal pelvic dilatation and postnatal sonographic features with the incidence of vesicoureteral reflux. MATERIALS AND METHODS: Maximum fetal renal pelvic dilatation was prospectively measured at a single center between 1990 and 2003. Dilatation 4 mm or greater at less than 33 weeks of gestation, or 7 mm or greater at more than 33 weeks was the threshold for inclusion in the study. Postnatal evaluation included ultrasound and voiding cystourethrogram. Postnatal data included vesicoureteral reflux incidence and grade, and caliceal and ureteral dilatation on ultrasound. RESULTS: Of 215 neonates 46 (21%) had vesicoureteral reflux. Mean renal pelvic dilatation was 14.4 mm in those with reflux, which was not statistically different than the mean of 11.8 mm in 169 with a normal voiding cystourethrogram. ROC analysis revealed that fetal renal pelvic dilatation was a poor discriminator of reflux. Reflux was identified in a significantly greater number of neonates with vs without postnatal calicectasis (20% vs 9%, p <0.05). When fetal renal pelvic dilatation was combined with postnatal calicectasis, only 5% of infants with dilatation less than 10 mm and isolated renal pelvic dilatation had reflux, whereas reflux was identified in 25% with fetal renal pelvic dilatation 10 mm or greater and calicectasis (p <0.02). CONCLUSIONS: The magnitude of fetal renal pelvic dilatation is not reliably predictive of reflux and this measure alone cannot be used to direct postnatal cystography. However, postnatal calicectasis appears to be an important predictor of vesicoureteral reflux in children with fetal renal pelvic dilatation. Expectant management can be considered in the subset of newborns with minimal dilatation (less than 10 mm) and absent calicectasis.

Prenatal diagnosis and neonatal management of congenital urethral diverticulum.

PURPOSE: We present a series of male neonates with prenatally detected anterior urethral diverticula and discuss postnatal management. MATERIALS AND METHODS: We retrospectively reviewed 3 cases of prenatally detected anterior urethral diverticula presenting between 1998 and 2005. RESULTS: An anterior urethral cystic mass was identified prenatally in all 3 patients. One mass spontaneously ruptured prenatally. The patient underwent diverticulectomy and urethroplasty at age 6 months. Two patients had obstructive uropathy with oligohydramnios and were delivered at 32 weeks of gestation. Both cases were managed by urethrostomy and subsequent diverticulectomy and urethroplasty at age 6 months. CONCLUSIONS: Infravesical obstruction from a urethral diverticulum can be accurately identified and differentiated from other causes prenatally. Marsupialization is an excellent option for temporary urinary diversion when the clinical situation precludes primary excision and repair.

The type and frequency of fetal renal disorders and management of renal pelvis dilatation.

OBJECTIVE: This study describes the frequency of sonographically detected fetal renal disorders, the correlation of fetal renal pelvis dilatation (RPD) with the need for postnatal surgery, and proposed management of RPD. METHODS: The study population consisted of 342 fetuses with prospectively identified isolated renal abnormalities and known follow-up. Fetuses with RPD were considered separately with respect to underlying renal disease, postnatal testing, and the need for surgical correction. Obstructive RPD was defined as the need for surgical treatment. Nonobstructive RPD included those cases that required no therapy. The correlation between prenatal RPD and the need for postnatal evaluation was examined. RESULTS: Renal pelvis dilatation was the primary postnatal sonographic finding in 66.4% of cases. The remainder were distributed between multicystic dysplastic kidney, duplication malformations, and reflux, with a smaller number of other diagnoses. Renal pelvis dilatation in the obstructive group was significantly greater than in the nonobstructive group. However, 10% of fetuses with maximum RPD of 10 mm or less had an obstructive process, whereas 58% of fetuses with RPD of greater than 10 mm did not have obstruction. There were no cases in which preterm delivery was necessitated by RPD. CONCLUSIONS: Renal pelvis dilatation is the most common fetal renal abnormality. The greater the RPD, the more likely it is due to obstruction. However, the overlap between obstruction and no obstruction dictates postnatal evaluation. In that RPD, regardless of degree, did not change the timing of delivery, a single follow-up sonographic examination either late in pregnancy or after delivery is considered adequate for follow-up of RPD detected earlier in pregnancy.

The magnitude of fetal renal pelvic dilatation can identify obstructive postnatal hydronephrosis, and direct postnatal evaluation and management.

PURPOSE: Up to 1% of prenatal ultrasounds will detect fetal renal pelvic dilatation. We sought to evaluate and determine whether fetal renal pelvic measurements may appropriately direct prenatal counseling and postnatal evaluation and management. MATERIALS AND METHODS: We performed a retrospective analysis of prospectively collected measurements of anteroposterior renal pelvic dilatation obtained at a single fetal maternal medicine center between 1990 and 2003. Fetuses with renal pelvic dilatation 4 mm or greater at less than 33 weeks of gestation, or 7 mm or greater at more than 33 weeks of gestation were evaluated postnatally at a single pediatric urology center. Infants with renal pelvic dilatation were evaluated with ultrasound, voiding cystourethrograms and renal scintigraphy. Renal obstruction was the main outcome measure assessed. Obstruction was defined as the need for surgery and was not based on the renal scan drainage time. Indications for surgery included declining function and increasing hydronephrosis. RESULTS: There were 257 neonates with prenatally detected renal pelvic dilatation. A mean maximum prenatal renal pelvic dilatation of 11.8 mm was seen in 195 patients with nonobstructive dilatation. In the 62 patients with obstruction there was a nearly 2-fold increase in the mean renal pelvic dilatation (22.3 mm), which was statistically significant. Receiver operating characteristic analysis revealed that when 15 mm renal pelvic dilatation is used as a threshold it correctly discriminates obstruction in at least 80% of fetuses with a sensitivity of 73% and a specificity of 82%. CONCLUSIONS: The magnitude of fetal renal pelvic dilatation is predictive of obstruction. Our results suggest that 15 mm renal pelvic dilatation represents a significant threshold. Receiver operating characteristic analysis provides a useful guide for prenatal counseling and may help to direct the postnatal evaluation.

Outcome analysis of prenatally detected ureteroceles associated with multicystic dysplasia.

PURPOSE: We assessed the outcome of a nonoperative approach in infants with prenatally detected ureteroceles associated with multicystic dysplasia. MATERIALS AND METHODS: A retrospective analysis of all infants with ureteroceles referred for evaluation of prenatally hydronephrosis was conducted. Ultrasound, cystogram and scintigraphy findings were tabulated. Clinical outcomes including infections and the need for surgical intervention were assessed. RESULTS: We identified 5 female and 3 male infants with prenatally detected ureteroceles and associated multicystic dysplasia. There were 4 cases with renal duplication and 4 with single systems. All infants were treated expectantly. One infant had a single urinary tract infection. The multicystic dysplastic moiety involuted by age 18 months in all children. The ureterocele collapsed in 3 children and remained stable in the remainder. None of the children required surgical intervention with a median and mean followup of 36 months (range 14 to 54). CONCLUSIONS: We identified a subset of prenatally diagnosed ureteroceles that had a benign clinical course and did not require surgical intervention with available followup. Careful evaluation and interpretation of postnatal studies will allow identification and observational management of this unique subset of ureteroceles associated with multicystic dysplasia and absence of hydroureteronephrosis.