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DOI 10.1542/peds.2019-1544

Медицинская библиотека · Pediatrics · 2020

Prenatal Repair of Myelomeningocele and School-age Functional Outcomes.

Houtrow Amy J, Thom Elizabeth A, Fletcher Jack M, Burrows Pamela K, Adzick N Scott, Thomas Nina H, Brock John W, Cooper Timothy, Lee Hanmin, Bilaniuk Larissa, Glenn Orit A, Pruthi Sumit, MacPherson Cora, Farmer Diana L, Johnson Mark P, Howell Lori J, Gupta Nalin, Walker William O

СтатьяМногоцентровое исследованиеРандомизированное исследованиеПоддержка NIH: внешняя программаПеревод в очереди

Аннотация

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.

Для цитирования: Houtrow Amy J, Thom Elizabeth A, Fletcher Jack M, Burrows Pamela K, Adzick N Scott, Thomas Nina H, Brock John W, Cooper Timothy, Lee Hanmin, Bilaniuk Larissa, Glenn Orit A, Pruthi Sumit, MacPherson Cora, Farmer Diana L, Johnson Mark P, Howell Lori J, Gupta Nalin, Walker William O. Prenatal Repair of Myelomeningocele and School-age Functional Outcomes.. Pediatrics. 2020. DOI: 10.1542/peds.2019-1544.

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