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Renal Replacement Therapy Rates Remain Stable Over Time For Children With Posterior Urethral Valves and Prune Belly Syndrome
Daryl McLeod1, Nicholas Beecroft2, Yuri Sebastião1, Christina Ching1, Brian Becknell1
1Nationwide Children's Hospital, Columbus, OH, 2The Ohio State University College of Medicine, Columbus, OH

Introduction
Posterior Urethral Valves (PUV) and Prune Belly Syndrome (PBS) are exceedingly rare yet encompass a disproportionate percentage of pediatric renal replacement therapy (RRT). Current literature on RRT risk in PUV and PBS represent single center experience. Despite evidence for increasing utilization of prenatal imaging and (in select centers) intervention, it is unknown if this has had any widespread effect on RRT rates. We aimed to investigate RRT risk, stratified by birth year and other patient factors in a large national sample.

Methods
Patients with PUV and PBS were identified in the Pediatric Health Information System (PHIS) database. Birth cohorts (admitted by third month of life) were developed from 37 freestanding US children′s hospitals where continuous hospital data (2006-2014) and at least 2 years of patient follow-up were available. Unconfirmed male sex (n=74) and death prior to discharge (n=81) was excluded. The primary outcome of interest was time from birth to RRT defined as dialysis or renal transplant. Predictor variables included birth cohort year, race, ethnicity, insurance, and diagnosis (PUV vs. PBS).

Results
In total 940 PUV and 142 PBS patients were identified. Median follow-up time was 4.7 years (Interquartile Range (IQR); 3.0-6.7). Overall 168 patients (14.7%) developed RRT with 132 (78.6%) requiring RRT within the first 2 years. On survival analysis we identified no difference in time to RRT stratifying by birth cohort years 2006-2012, race, ethnicity, insurance or diagnosis.

Conclusions
This is the first known study to evaluate the risk of RRT in children with PUV or PBS on a national level. The majority of patients who required RRT did so in the first 2 years of life. This is consistent with single center data indicating a bimodal time to RRT with the second peak occurring at puberty. Our study was not designed to capture the expected second peak of progression. Interestingly we also found no difference in the time to RRT by birth cohort year. The stability of time to RRT over our study period exemplifies the difficulties in modifying the natural history of PUV and PBS, despite continued technological advancements. Future studies need to focus on improved risk stratification and potential interventions for those children who escape the first 2 years of life with intact renal function, as they may have a greater potential for disease course modification.


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