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胎儿或产后手术后伴有脊髓脊膜膨出的儿童的神经外科手术:一项比较效果研究。

Neurosurgical procedures for children with myelomeningocele after fetal or postnatal surgery: a comparative effectiveness study.

机构信息

Division of Pediatric Neurology and Developmental Medicine, Department of Pediatrics, Duke University Medical Center, Durham, NC, USA.

Division of Neonatology, Department of Pediatrics, Duke University Medical Center, Durham, NC, USA.

出版信息

Dev Med Child Neurol. 2021 Nov;63(11):1294-1301. doi: 10.1111/dmcn.14792. Epub 2021 Jan 2.

Abstract

AIM

To compare the frequencies of neurosurgical procedures to treat comorbid conditions of myelomeningocele in patients who underwent fetal surgery versus postnatal surgery for closure of the placode.

METHOD

By utilizing the National Spina Bifida Patient Registry in a comparative effectiveness study, 298 fetal surgery patients were matched by birthdate (±3mo) and spina bifida clinic site with one to three postnatal surgery patients (n=648). Histories were obtained by record review on enrollment and yearly subsequently. Multivariable Poisson regression was used to compare frequencies of procedures between cohorts, with adjustments for sex, ethnicity, insurance status, spinal segmental level of motor function, age at last visit recorded in the Registry, and, for shunt revision in shunted patients, age at cerebrospinal fluid (CSF) diversion.

RESULTS

The median age at last visit was 4 years. In fully adjusted analyses in patients aged at least 12 months old, fetal surgery was associated with decreased frequency of CSF diversion for hydrocephalus by ventriculoperitoneal shunt insertion or endoscopic third ventriculostomy compared with postnatal surgery (46% vs 79%; incidence rate ratio=0.61; 95% confidence interval [CI] 0.53-0.71; p<0.01). Over all ages, fetal surgery was associated with decreased frequency of Chiari decompression for brainstem dysfunction (3% vs 7%; incidence rate ratio=0.41; 95% CI 0.19-0.88; p=0.02). Also over all ages, differences were not significant in frequencies of shunt revision in shunted patients (53% vs 55%; incidence rate ratio=0.87; 95% CI 0.69-1.11; p=0.27), nor tethered cord release for acquired spinal cord dysfunction (18% vs 16%; incidence rate ratio=1.11; 95% CI 0.84-1.47; p=0.46).

INTERPRETATION

Even with the variations inherent in clinical practice, fetal surgery was associated with lower frequencies of CSF diversion and of Chiari decompression, independent of covariates. What this paper adds Fetal surgery was associated with lower frequencies of cerebrospinal fluid diversion and decompression of Chiari II malformation than postnatal surgery. Frequencies of ventriculoperitoneal shunt revision and tethered cord release were not significantly different between cohorts.

摘要

目的

通过利用全国脊髓脊膜膨出患者注册中心的一项比较效果研究,比较胎儿手术和产后手术治疗脊膜膨出患者合并症的神经外科手术频率,这些患者的脊膜膨出均已通过手术进行了闭合。

方法

通过利用全国脊髓脊膜膨出患者注册中心的一项比较效果研究,通过记录审查,以出生日期(±3 个月)和脊膜膨出诊所为基础,将 298 名胎儿手术患者与 1 至 3 名产后手术患者(n=648)进行匹配。在登记时和随后每年获取病史。采用多变量泊松回归比较队列之间的手术频率,调整了性别、种族、保险状况、运动功能脊髓节段水平、记录在注册处的最后就诊年龄,以及分流患者的分流管修复时的脑脊液(CSF)引流年龄。

结果

中位最后就诊年龄为 4 岁。在至少 12 个月大的患者的完全调整分析中,与产后手术相比,胎儿手术与脑室腹膜分流术或内镜第三脑室造瘘术行脑积水 CSF 分流的频率降低有关(46% vs 79%;发病率比=0.61;95%置信区间[CI]0.53-0.71;p<0.01)。在所有年龄段,胎儿手术与脑桥功能障碍行 Chiari 减压的频率降低有关(3% vs 7%;发病率比=0.41;95%CI0.19-0.88;p=0.02)。同样在所有年龄段,分流患者的分流管修复频率(53% vs 55%;发病率比=0.87;95%CI0.69-1.11;p=0.27)和获得性脊髓功能障碍行脊髓松解术的频率(18% vs 16%;发病率比=1.11;95%CI0.84-1.47;p=0.46)差异均无统计学意义。

结论

即使存在临床实践中的固有差异,胎儿手术与 CSF 分流和 Chiari 减压的频率降低相关,且不受协变量影响。本文的补充内容胎儿手术与产后手术相比,CSF 分流和 Chiari II 畸形减压的频率较低。两组患者的脑室腹膜分流管修复和脊髓松解术的频率无显著差异。

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