The Management of Myelomeningocele Study: full cohort 30-month pediatric outcomes

Farmer, Diana L.; Thom, Elizabeth; Brock, John W.; Burrows, Pamela K.; Johnson, Mark P.; Howell, Lori J.; Farrell, Jody A.; Gupta, Nalin; Adzick, N. Scott

doi:10.1016/j.ajog.2017.12.001

Cited by 218 publications

(239 citation statements)

References 18 publications

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“…The location of the upper level of the spina bifida defect was between L1 and L5 in the majority of cases in both groups (70.8% in the prenatal group vs 70.5% in the postnatal group; P = 0.2). A difference between motor function and anatomical level of the lesion was reported only by Farmer et al ,. who observed that children who underwent prenatal repair were significantly more likely to have motor function at least two levels better than expected based on the anatomical level of the defect, compared with those who were operated on postnatally (26.4% vs 11.4%; P < 0.02).…”

Section: Resultsmentioning

confidence: 91%

“…According to the Cochrane risk of bias tool, the included RCT had low risk of bias and could thus be defined as being high quality. Risk of bias arising from the randomization process was low, as a secure website was used for randomization stratified by fetal surgery center.…”

Section: Resultsmentioning

confidence: 99%

“…Analysis of the 30‐month outcome of the full randomized MOMS cohort demonstrated comparable neurodevelopmental impairment rates in children operated on prenatally with (7/39 (17.9%)) or without (4/48 (8.3%); relative risk, 0.90 (95% CI, 0.75–1.06); P = 0.21) a shunt. However, the number of patients was limited, and similar outcome data were not available for the postnatal group.…”

Section: Discussionmentioning

confidence: 98%

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Neurodevelopmental outcome of children with spina bifida aperta repaired prenatally vs postnatally: systematic review and meta‐analysis

Inversetti

Veeken

Thompson

et al. 2019

Ultrasound in Obstet & Gyne

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Objective To assess the neurodevelopmental outcome of children with spina bifida aperta (SBA) treated prenatally as compared to those treated postnatally. Methods We performed a systematic review of the literature in PubMed/MEDLINE, EMBASE, Web of Science and The Cochrane Library, comparing the neurological outcome of infants with SBA treated prenatally vs postnatally. Only randomized controlled trials (RCTs) and non‐randomized prospective controlled studies were included. The primary outcome assessed was neurodevelopmental impairment at the age of 1 year or later. Secondary outcomes were preterm birth, need for ventriculoperitoneal (VP) shunt by 12 months of age, absence of signs of hindbrain herniation at the first postnatal magnetic resonance imaging (MRI) evaluation and independent ambulation evaluated at 30 months. Results Of 11 359 studies identified through the electronic search, six met the inclusion criteria and were assessed in full text and two, one RCT and one prospective cohort study, were ultimately included in the meta‐analysis. Sensitivity analysis did not show any difference between the outcomes of the RCT alone and those of the pooled RCT and prospective cohort study. This allowed neurodevelopmental assessment of 213 children between 14 and 53 months of age. Neurodevelopment was assessed by the Bayley Scales of Infant Development II (BSID‐II) mental development index corrected for chronological age, with a cut‐off of ≥ 70 (representing no more than 2 SD below the mean). The presence of neurodevelopmental impairment was similar between children who underwent prenatal (25/105 (23.8%)) and those who had postnatal (30/108 (27.8%)) repair of SBA (odds ratio (OR), 0.82 (95% CI, 0.43–1.56); P = 0.54), although the risk of prematurity was higher in the prenatal‐repair group (OR, 17.62 (95% CI, 7.60–40.87); P < 0.0001). For every two fetuses operated on before birth, there was, compared with those operated on after birth, one additional premature birth (number needed to harm = 2 (95% CI, 1–3)). The need for VP shunt placement by 12 months of age was lower in the prenatal‐repair group (45/109 (41.3%)) than in children that had postnatal repair (93/112 (83.0%); OR, 0.14 (95% CI, 0.08–0.26); P < 0.0001). Data on neurodevelopmental impairment in children with a shunt were available only for patients from the prenatal‐surgery group of the RCT; in this subgroup, the likelihood for impairment was similar between children who did (7/39 (17.9%)) and those who did not (4/48 (8.3%)) have shunt placement (P = 0.21). At first postnatal MRI evaluation, no signs of hindbrain herniation were detected in 28/88 (31.8%) children who were operated on prenatally compared with 4/89 (4.5%) who had postnatal repair (OR, 9.45 (95% CI, 3.12–28.64); P < 0.0001). Independent ambulation at 30 months was achieved by 41/109 (37.6%) children who underwent prenatal repair compared with 21/111 (18.9%) who had postnatal repair (OR, 2.59 (95% CI, 1.39–4.86); P = 0.003). Conclusion The risk of neurodevelopmental impairment in infan...

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Section: Resultsmentioning

confidence: 91%

Section: Resultsmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 98%

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Neurodevelopmental outcome of children with spina bifida aperta repaired prenatally vs postnatally: systematic review and meta‐analysis

Inversetti

Veeken

Thompson

et al. 2019

Ultrasound in Obstet & Gyne

View full text Add to dashboard Cite

show abstract

“…However, when there are only two studies being combined, the potential for bias from a large loss to follow‐up remains exposed. While there was a comparable rate of prematurity in the cohort study, compared with the MOMS trial, neurological impairment was higher, the rate of independent ambulation was lower and complete reversal of hindbrain herniation was lower. The only improvement was that the shunt rate in prenatally repaired infants was lower.…”

mentioning

confidence: 77%

Management of prenatally diagnosed spina bifida: how do we move ahead?

Bebbington

2019

Ultrasound in Obstet & Gyne

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“…Subsequent analysis of the full MOMS cohort not only validated the initial results, but also identified additional risk factors of adverse outcomes . Tulipan et al demonstrated that ventricular dimension at the time of fMMC surgery affects the need for ventriculoperitoneal shunting and suggested that there is limited benefit for patients with ventricle measurements >15mm (normal <10mm).…”

Section: Momsmentioning

confidence: 99%

Fetal surgical intervention for myelomeningocele: lessons learned, outcomes, and future implications

Danzer

Joyeux

Flake

et al. 2019

Develop Med Child Neuro

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Fetal myelomeningocele (fMMC) closure (spina bifida aperta) has become a care option for patients that meet inclusion criteria, but it is clear that fetal intervention, while improving outcomes, is not a cure. This review will: (1) focus on the rationale for fMMC surgery based on preclinical studies and observations that laid the foundation for human pilot studies and a randomized controlled trial; (2) summarize important clinical outcomes; (3) discuss the feasibility, efficacy, and safety of recent developments in fetal surgical techniques and approaches; and (4) highlight future research directions. Given the increased risk of maternal and fetal morbidity associated with prenatal intervention, accompanied by the increasing number of centres performing interventions worldwide, teams involved in the care of these patients need to proceed with caution to maintain technical expertise, competency, and patient safety. Ongoing assessment of durability of the benefits of fMMC surgery, as well as additional refinement of patient selection criteria and counselling, is needed to further improve outcomes and reduce the risks to the mother and fetus. What this paper adds High‐quality prospective studies are needed to broaden the indication for fetal surgery in the general myelomeningocele population. Innovative minimally invasive approaches have had promising results, yet lack comprehensive and robust experimental or clinical evaluation. Important information to help families make informed decisions regarding fetal surgery for myelomeningocele is provided.

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The Management of Myelomeningocele Study: full cohort 30-month pediatric outcomes

Cited by 218 publications

References 18 publications

Neurodevelopmental outcome of children with spina bifida aperta repaired prenatally vs postnatally: systematic review and meta‐analysis

Neurodevelopmental outcome of children with spina bifida aperta repaired prenatally vs postnatally: systematic review and meta‐analysis

Management of prenatally diagnosed spina bifida: how do we move ahead?

Fetal surgical intervention for myelomeningocele: lessons learned, outcomes, and future implications

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