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Original Investigation
February 8, 2021

Prenatal Repair and Physical Functioning Among Children With Myelomeningocele: A Secondary Analysis of a Randomized Clinical Trial

Author Affiliations
  • 1Department of Physical Medicine & Rehabilitation, University of Pittsburgh, Pittsburgh, Pennsylvania
  • 2Children’s Hospital of Pittsburgh, University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania
  • 3The Biostatistics Center, Milken Institute of Public Health, George Washington University, Washington, DC
  • 4Department of Physical Therapy, Jefferson University, Philadelphia, Pennsylvania
  • 5Department of Physical Therapy, Fresno State University, Fresno, California
  • 6Department of Physical Therapy, Vanderbilt University, Nashville, Tennessee
  • 7Department of Surgery, Children’s Hospital of Philadelphia, Philadelphia, Pennsylvania
  • 8Department of Psychology, University of Houston, Houston, Texas
  • 9Department of Neurosurgery, University of California, San Francisco, San Francisco
  • 10Center for Fetal Diagnosis and Treatment, Children’s Hospital of Philadelphia, Philadelphia, Pennsylvania
  • 11Department of Surgery, Vanderbilt University, Nashville, Tennessee
  • 12Department of Surgery, University of California, San Francisco, San Francisco
  • 13Department of Pediatrics, University of Washington, Seattle
JAMA Pediatr. 2021;175(4):e205674. doi:10.1001/jamapediatrics.2020.5674
Key Points

Question  Do children who have undergone prenatal repair of myelomeningocele have better functional mobility and motor levels than school-aged children who have had standard postnatal repair?

Findings  In this secondary analysis of 154 children from the Management of Myelomeningocele Study randomized clinical trial, 51.3% of children who underwent prenatal repair could independently walk community distances compared with 23.1% of children who underwent standard postnatal repair. Children in the prenatal repair group were less likely to have a motor function level worse than their anatomic lesion level (adjusted relative risk, 0.44).

Meaning  These findings suggest that functional mobility and motor levels are improved for children who underwent prenatal myelomeningocele repair vs standard postnatal repair.

Abstract

Importance  The Management of Myelomeningocele Study (MOMS), a randomized clinical trial of prenatal vs standard postnatal repair for myelomeningocele, found that prenatal repair reduced hydrocephalus and hindbrain herniation and improved motor function in children aged 12 to 30 months. The Management of Myelomeningocele Study Follow-up (MOMS2) was conducted in children at ages 5 to 10 years. The primary (neurocognitive) outcome has already been reported.

Objective  To determine whether MOMS2 participants who had prenatal repair have better physical functioning than those with postnatal repair.

Design, Setting, and Participants  Participants from MOMS were recruited for participation in the follow-up study, MOMS2, conducted from April 9, 2012, to April 15, 2017. For this secondary analysis of the randomized clinical trial, trained examiners without knowledge of the treatment group evaluated the physical characteristics, self-care skills, neurologic function, and mobility of the children. Physical functioning outcomes were compared between the prenatal and postnatal repair groups.MOMS2 was conducted at the same 3 clinical sites as MOMS. Home visits were conducted for families who were unable to travel to one of the clinical sites.Of the 161 children with myelomeningocele aged 5 to 10 years old enrolled in MOMS2, 154 had a physical examination and were included in the analyses.

Exposures  Prenatal repair of myelomeningocele.

Main Outcomes and Measures  Prespecified secondary trial outcomes of self-care skills, functional mobility, walking skills, and motor level.

Results  This analysis included 78 children with postnatal repair (mean [SD] age, 7.4 [2.1] years; 50 girls [64.1%]; 69 White children [88.5%]) and 76 with prenatal repair (mean [SD] age, 7.5 [1.2] years; 43 boys [56.6%]; 70 White children [92.1%]). Children in the prenatal repair group were more competent with self-care skills (mean [SD] percentage of maximum FRESNO Scale score, 90.8% [9.6%] vs 85.5% [17.6%]) and were commonly community ambulators per the Modified Hoffer Classification (51.3% prenatal vs 23.1% postnatal; adjusted relative risk [aRR] for sex, 1.70; 95% CI, 1.23-2.34). Children with prenatal repair also performed the 10-m walk test 1 second faster (difference in medians, 1.0; 95% CI, 0.3-1.7), had better gait quality (adjusted mean difference for home distances of 5 m, 1.71; 95% CI, 1.14-2.54), and could perform higher-level mobility skills (adjusted mean difference for motor total, 5.70; 95% CI, 1.97-11.18). Children in the prenatal repair group were less likely to have a motor function level worse than their anatomic lesion level (aRR, 0.44; 95% CI, 0.25-0.77).

Conclusions and Relevance  This secondary analysis of a randomized clinical trial found that the physical functioning benefits of prenatal repair for myelomeningocele reported at age 30 months persisted into school age. These findings indicate the benefit of prenatal repair of myelomeningocele for school-aged children.

Trial Registration  ClinicalTrials.gov Identifier: NCT00060606.

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