Reversal of hindbrain herniation after maternal-fetal surgery for myelomeningocele subsequently impacts on brain stem function

E. Danzer, R. S. Finkel, N. E. Rintoul, M. W. Bebbington, E. S. Schwartz, D. M. Zarnow, N. S. Adzick, M. P. Johnson

Research output: Contribution to journalArticle

50 Scopus citations

Abstract

The aim of our study was to delineate whether the reversal of hindbrain herniation (HH) following fetal myelomeningocele (fMMC) closure subsequently reduces the incidence and severity of HH-associated brainstem dysfunction (BSD). Prior to the NIH-sponsored Management of Myelomeningocele Study (MOMS) trial, 54 children underwent fMMC closure at our institution. Forty-eight (89%) families participated in a structured survey focusing on HH-associated BSD (e.g., apnea, neurogenic dysphagia (ND), gastro-esophageal reflux disease [GERD], neuro-ophthalmologic disturbances [NOD]). Median age at follow-up was 72 months (range: 46-98). Fifty-percent required shunting. HH-related symptoms were completely absent in 15 (63%) non-shunted and 10 (42%) shunted children (P=0.15). No HH-related death occurred and none developed severe persistent cyanotic apnea. ND was reported in 2 (8%) non-shunted and 9 (38%) shunted infants (P=0.03). Mild GERD (medically managed) developed in 2 (8%) without and 6 (25%) with shunt placement (P=0.24). NOD was found in 6 (25 %) and 13 (54 %) of nonshunted and shunted children, respectively (P=0.07). The majority of fMMC children developed no or only mild BSD at follow-up. Our data support the hypothesis that neurodevelopmental deficits associated with MMC are at least partially acquired and that reversal of HH following fMMC surgery may help to reduce the incidence and severity of BSD.

Original languageEnglish
Pages (from-to)359-362
Number of pages4
JournalNeuropediatrics
Volume39
Issue number6
DOIs
StatePublished - Dec 1 2008
Externally publishedYes

Keywords

  • Apnea
  • Brain stem dysfunction
  • Fetal surgery
  • Hindbrain herniation
  • Myelomeningocele

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    Danzer, E., Finkel, R. S., Rintoul, N. E., Bebbington, M. W., Schwartz, E. S., Zarnow, D. M., Adzick, N. S., & Johnson, M. P. (2008). Reversal of hindbrain herniation after maternal-fetal surgery for myelomeningocele subsequently impacts on brain stem function. Neuropediatrics, 39(6), 359-362. https://doi.org/10.1055/s-0029-1202835