Prenatal Repair of Myelomeningocele and School-age Functional Outcomes

被引:95
|
作者
Houtrow, Amy J. [1 ]
Thom, Elizabeth A. [2 ]
Fletcher, Jack M. [3 ]
Burrows, Pamela K. [2 ]
Adzick, N. Scott [4 ]
Thomas, Nina H. [4 ]
Brock, John W., III [5 ]
Cooper, Timothy [5 ]
Lee, Hanmin [6 ]
Bilaniuk, Larissa [4 ]
Glenn, Orit A. [6 ]
Pruthi, Sumit [5 ]
MacPherson, Cora [2 ]
Farmer, Diana L. [7 ,8 ]
Johnson, Mark P. [4 ]
Howell, Lori J. [4 ]
Gupta, Nalin [6 ]
Walker, William O. [9 ]
机构
[1] Univ Pittsburgh, Dept Phys Med & Rehabil, Pittsburgh, PA USA
[2] George Washington Univ, Biostat Ctr, Sch Publ Hlth, Milken Inst, Washington, DC USA
[3] Univ Houston, Dept Psychol, Houston, TX USA
[4] Childrens Hosp Philadelphia, Philadelphia, PA 19104 USA
[5] Vanderbilt Univ, Med Ctr, Nashville, TN USA
[6] Univ Calif San Francisco, Dept Surg, San Francisco, CA USA
[7] Univ Calif Davis, Dept Neurol Surg, Davis, CA 95616 USA
[8] Univ Calif Davis, Dept Pediat, Davis, CA 95616 USA
[9] Seattle Childrens Hosp, Seattle, WA USA
基金
美国国家卫生研究院;
关键词
CHIARI-II MALFORMATION; FETAL SURGERY; SPINA-BIFIDA; MOBILITY; CHILDREN; BIRTH;
D O I
10.1542/peds.2019-1544
中图分类号
R72 [儿科学];
学科分类号
100202 ;
摘要
BACKGROUND AND OBJECTIVES: The Management of Myelomeningocele Study (MOMS), a randomized trial of prenatal versus postnatal repair for myelomeningocele, found that prenatal surgery resulted in reduced hindbrain herniation and need for shunt diversion at 12 months of age and better motor function at 30 months. In this study, we compared adaptive behavior and other outcomes at school age (5.9-10.3 years) between prenatal versus postnatal surgery groups. METHODS: Follow-up cohort study of 161 children enrolled in MOMS. Assessments included neuropsychological and physical evaluations. Children were evaluated at a MOMS center or at a home visit by trained blinded examiners. RESULTS: The Vineland composite score was not different between surgery groups (89.0 +/- 9.6 in the prenatal group versus 87.5 +/- 12.0 in the postnatal group; P = .35). Children in the prenatal group walked without orthotics or assistive devices more often (29% vs 11%; P = .06), had higher mean percentage scores on the Functional Rehabilitation Evaluation of Sensori-Neurologic Outcomes (92 +/- 9 vs 85 +/- 18; P < .001), lower rates of hindbrain herniation (60% vs 87%; P < .001), had fewer shunts placed for hydrocephalus (49% vs 85%; P < .001) and, among those with shunts, fewer shunt revisions (47% vs 70%; P = .02) than those in the postnatal group. Parents of children repaired prenatally reported higher mean quality of life z scores (0.15 +/- 0.67 vs 0.11 +/- 0.73; P = .008) and lower mean family impact scores (32.5 +/- 7.8 vs 37.0 +/- 8.9; P = .002). CONCLUSIONS: There was no significant difference between surgery groups in overall adaptive behavior. Long-term benefits of prenatal surgery included improved mobility and independent functioning and fewer surgeries for shunt placement and revision, with no strong evidence of improved cognitive functioning.
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页数:9
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