Prenatal Myelomeningocele Closure Is Associated with Higher Continence And Lower Rate of Clean Intermittent Catheterization - A National Spina Bifida Patient Registry Study
Hsin-Hsiao Scott Wang, MD, MPH, MBAn1, Tanya Logvinenko, PhD1, Benjamin Warf, MD1, Lawrence Karlin, MD1, Stuart Bauer, MD1, Erin McNamara, MD1, John Wiener, MD2, David Chu, MD, MSc3, Robin Bowman, MD3, Charles Rose, PhD4, Tiebin Liu, PhD4, Heidi Castillo, MD5, Jonathan Castillo, MD5, Andrew Foy, MD6, Kathryn Smith, MD7, Alexander Van Speybroack, MD7, Carlos Estrada, MD, MBA1.
1Boston Children's Hospital, Boston, MA, USA, 2Duke University Medical Center, Durham, NC, USA, 3Lurie Children's Hospital of Chicago, Chicago, IL, USA, 4Centers for Disease Control and Prevention, Atlanta, GA, USA, 5Texas Children's Hospital, Houston, TX, USA, 6Medical College of Wisconsin, Milwaukee, WI, USA, 7Children's Hospital Los Angeles, Los Angeles, CA, USA.
Introduction: Although prenatal closure of myelomeningocele (MMC) has had demonstrable benefits, its long-term impact on urologic outcomes is not well known. We compared urologic outcomes between children with prenatal and those with postnatal MMC repair in a large national registry, the National Spina Bifida Patient Registry (NSBPR).
Methods: We included children born during 1990 through 2021 who had prenatal MMC closure and were followed in the NSBPR and compared their urologic outcomes with those in children from the same period with MMC closed postnatally. The outcomes compared were time from birth to clean intermittent catheterization (CIC), to voiding volition without incontinence (defined as voiding & leaks equal or less than once a month), and to urinary continence (defined as voiding/CIC & leaks equal or less than once a month). Covariates included year of birth, sex, race, and functional level of neurologic lesion. The overall and the probability over time of these outcomes were estimated by fitting multivariable Cox proportional hazard models with interval censoring.
Results: We identified 474 children with prenatal and 6,406 children with postnatal MMC closures. Among the 6% prenatal and 5% postnatal patients who reached voiding volition without incontinence, it happened at median age 8.0 (Interquartile Range [IQR] 4.7-12.5) years and 11.0 (IQR 6.5-16.1) years respectively (Figure 1). Among the 30% of prenatal patients and 42% of postnatal patients who reached urinary continence, the median age was 9 (IQR 4.0-14.2) years and 11.8 (IQR 7.3-16.8) years, respectively (Figure 2). After adjusting for race, sex, and functional level of neurologic lesion, the respective hazards of reaching voiding volition without incontinence and urinary continence were 80% higher (Hazard Ratio [HR] 1.8, 95% CI 1.3-2.5, p<0.001) and 60% higher (HR 1.6, 95% CI 1.4-2.0, p<0.001) for patients who had prenatal closure surgery compared with individuals who had postnatal closure. 6495 patients with spina bifida (461 with prenatal and 5934 with postnatal procedures) had CIC data in NSBPR. Among 38% and 56% of those whose MMC was closed prenatally and postnatally, respectively, CIC was initiated at a median age of 1.1 (IQR 0.5-4.5) years in the prenatal closure group and 1.6 (IQR 0.5-4.5) years in the postnatal closure group (p<0.001). After adjusting for race, sex, and functional level of neurologic lesion, the overall average hazard of starting CIC for those with postnatal MMC closure was 30% higher (HR 1.3, 95%CI 1.2-1.5, p<0.001) than those in the prenatal closure group. CONCLUSIONS: Compared with postnatal closure, prenatal MMC closure was associated with lower hazard of requiring CIC and higher chance of achieving voiding volition without incontinence and urinary continence. The increased probability of favorable urologic outcomes among patients with prenatal MMC closure could provide useful insights for spina bifida patient care and counseling.
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