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Randomized Controlled Trial
. 2021 Apr 1;175(4):e205674.
doi: 10.1001/jamapediatrics.2020.5674. Epub 2021 Apr 5.

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

Amy J Houtrow  1   2 , Cora MacPherson  3 , Janet Jackson-Coty  4 , Monica Rivera  5 , Laura Flynn  6 , Pamela K Burrows  3 , N Scott Adzick  7 , Jack Fletcher  8 , Nalin Gupta  9 , Lori J Howell  10 , John W Brock 3rd  11 , Hanmin Lee  12 , William O Walker  13 , Elizabeth A Thom  3
Affiliations
Randomized Controlled Trial

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

Amy J Houtrow et al. JAMA Pediatr. .

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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Conflict of interest statement

Conflict of Interest Disclosures: None reported.

Figures

Figure.
Figure.. Flow Diagram for Enrollment and Follow-up
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