Sasidhar Karuparti, Tracy M Flanders, Ashley Dunbar, Kaamya Varagur, Jennifer M Strahle
{"title":"接受产前和产后手术治疗的脊髓脊膜膨出症患者的头部发育情况。","authors":"Sasidhar Karuparti, Tracy M Flanders, Ashley Dunbar, Kaamya Varagur, Jennifer M Strahle","doi":"10.3171/2023.11.PEDS23328","DOIUrl":null,"url":null,"abstract":"<p><strong>Objective: </strong>The need for permanent CSF diversion is lower in patients who have undergone prenatal surgery for myelomeningocele (MMC) than in those who have undergone postnatal closure. Differences in brain development and head growth between treatment groups are not known, particularly for those who do not require surgical treatment for hydrocephalus. The objective of this study was to determine differences in head growth and to generate MMC-specific head circumference (HC) growth curves for patients who underwent either prenatal or postnatal surgery.</p><p><strong>Methods: </strong>The authors retrospectively identified patients from St. Louis Children's Hospital who were treated for MMC between 2016 and 2021. HC data were obtained from birth until the most recent follow-up or hydrocephalus treatment. Nonlinear least-squares regression analysis was performed to fit the data into four models: two-term power, Gompertz, West ontogenetic, and Weibull. Subsequently, the curves were assessed for their utility in predicting hydrocephalus treatment.</p><p><strong>Results: </strong>Sixty-one patients (29 females [47.5%], 25 [41%] underwent prenatal surgery, mean gestational age at birth 36.6 weeks) were included in the study. The Weibull model best fit the HC data (prenatal adjusted R2 = 0.95, postnatal adjusted R2 = 0.95), while the Gompertz model had the worst fit (prenatal adjusted R2 = 0.56, postnatal adjusted R2 = 0.39) across both cohorts. Prenatal MMC repair patients had significantly larger HC measurements than their postnatal repair counterparts. The 50th percentile of the Weibull curve was determined as a useful threshold for hydrocephalus treatment: children with HC measurements that crossed and remained above this threshold were significantly more likely to have hydrocephalus treatment regardless of time of MMC repair (prenatal relative risk [RR] 10.0 [95% CI 1.424-70.220], sensitivity 85.7% [95% CI 0.499-0.984], and specificity 82.4% [95% CI 0.600-0.948]; postnatal RR 4.750 [95% CI 1.341-16.822], sensitivity 90.5% [95% CI 0.728-0.980], and specificity 75.0% [95% CI 0.471-0.924]). The HC growth curves of the MMC patients treated prenatally were significantly larger than the WHO HC curves (p < 0.001).</p><p><strong>Conclusions: </strong>The Weibull model was identified as the HC growth curve with the best fit for MMC patients and serves as a useful predictor of hydrocephalus treatment. For MMC patients with hydrocephalus, prenatal repair patients fit the model well but postnatal repair patients did not, potentially indicating different mechanisms of hydrocephalus development. Those treated prenatally had significantly larger HC measurements compared with both the general population and those treated postnatally. Further study is needed to understand the long-term cognitive outcomes and optimal management of clinically asymptomatic patients with large HC measurements who were treated prenatally for MMC.</p>","PeriodicalId":16549,"journal":{"name":"Journal of neurosurgery. 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The objective of this study was to determine differences in head growth and to generate MMC-specific head circumference (HC) growth curves for patients who underwent either prenatal or postnatal surgery.</p><p><strong>Methods: </strong>The authors retrospectively identified patients from St. Louis Children's Hospital who were treated for MMC between 2016 and 2021. HC data were obtained from birth until the most recent follow-up or hydrocephalus treatment. Nonlinear least-squares regression analysis was performed to fit the data into four models: two-term power, Gompertz, West ontogenetic, and Weibull. Subsequently, the curves were assessed for their utility in predicting hydrocephalus treatment.</p><p><strong>Results: </strong>Sixty-one patients (29 females [47.5%], 25 [41%] underwent prenatal surgery, mean gestational age at birth 36.6 weeks) were included in the study. The Weibull model best fit the HC data (prenatal adjusted R2 = 0.95, postnatal adjusted R2 = 0.95), while the Gompertz model had the worst fit (prenatal adjusted R2 = 0.56, postnatal adjusted R2 = 0.39) across both cohorts. Prenatal MMC repair patients had significantly larger HC measurements than their postnatal repair counterparts. The 50th percentile of the Weibull curve was determined as a useful threshold for hydrocephalus treatment: children with HC measurements that crossed and remained above this threshold were significantly more likely to have hydrocephalus treatment regardless of time of MMC repair (prenatal relative risk [RR] 10.0 [95% CI 1.424-70.220], sensitivity 85.7% [95% CI 0.499-0.984], and specificity 82.4% [95% CI 0.600-0.948]; postnatal RR 4.750 [95% CI 1.341-16.822], sensitivity 90.5% [95% CI 0.728-0.980], and specificity 75.0% [95% CI 0.471-0.924]). The HC growth curves of the MMC patients treated prenatally were significantly larger than the WHO HC curves (p < 0.001).</p><p><strong>Conclusions: </strong>The Weibull model was identified as the HC growth curve with the best fit for MMC patients and serves as a useful predictor of hydrocephalus treatment. For MMC patients with hydrocephalus, prenatal repair patients fit the model well but postnatal repair patients did not, potentially indicating different mechanisms of hydrocephalus development. Those treated prenatally had significantly larger HC measurements compared with both the general population and those treated postnatally. Further study is needed to understand the long-term cognitive outcomes and optimal management of clinically asymptomatic patients with large HC measurements who were treated prenatally for MMC.</p>\",\"PeriodicalId\":16549,\"journal\":{\"name\":\"Journal of neurosurgery. 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引用次数: 0
摘要
目的:产前接受过脊髓膜膨出(MMC)手术的患者对永久性脑脊液转流的需求低于产后接受闭合手术的患者。治疗组之间大脑发育和头部生长的差异尚不清楚,尤其是那些不需要手术治疗脑积水的患者。本研究的目的是确定接受产前或产后手术的患者在头部生长方面的差异,并生成特定的 MMC 头围(HC)生长曲线:作者回顾性地确定了圣路易斯儿童医院在2016年至2021年间接受MMC治疗的患者。从出生到最近一次随访或脑积水治疗期间的HC数据均已获得。进行了非线性最小二乘回归分析,将数据拟合为四种模型:两期幂模型、冈珀茨模型、West ontogenetic模型和Weibull模型。随后,评估了这些曲线在预测脑积水治疗中的实用性:研究共纳入 61 名患者(29 名女性[47.5%],25 名[41%]接受了产前手术,出生时平均胎龄为 36.6 周)。Weibull模型最适合HC数据(产前调整R2 = 0.95,产后调整R2 = 0.95),而Gompertz模型在两个组群中的拟合效果最差(产前调整R2 = 0.56,产后调整R2 = 0.39)。产前 MMC 修复患者的 HC 测量值明显大于产后修复患者。Weibull 曲线的第 50 百分位数被确定为治疗脑积水的有用阈值:无论 MMC 修复时间长短,HC 测量值超过并保持在该阈值以上的患儿接受脑积水治疗的可能性明显更高(产前相对风险 [RR] 10.0 [95% CI 1.424-70.220],敏感性 85.7% [95% CI 0.499-0.984],特异性 82.4% [95% CI 0.600-0.948];出生后 RR 4.750 [95% CI 1.341-16.822],敏感性 90.5% [95% CI 0.728-0.980],特异性 75.0% [95% CI 0.471-0.924])。产前接受治疗的 MMC 患者的 HC 生长曲线明显大于 WHO HC 曲线(P < 0.001):Weibull模型被认为是最适合MMC患者的HC生长曲线,是预测脑积水治疗的有用指标。对于伴有脑积水的 MMC 患者,产前修复的患者与模型的拟合度较高,而产后修复的患者与模型的拟合度较低,这可能表明脑积水的发展机制不同。与普通人群和产后接受治疗的患者相比,产前接受治疗的患者的脑积水测量值明显增大。要了解临床无症状、HC 测量值较大且在产前接受过 MMC 治疗的患者的长期认知结果和最佳治疗方法,还需要进一步的研究。
Head growth in patients with myelomeningocele treated with prenatal and postnatal surgery.
Objective: The need for permanent CSF diversion is lower in patients who have undergone prenatal surgery for myelomeningocele (MMC) than in those who have undergone postnatal closure. Differences in brain development and head growth between treatment groups are not known, particularly for those who do not require surgical treatment for hydrocephalus. The objective of this study was to determine differences in head growth and to generate MMC-specific head circumference (HC) growth curves for patients who underwent either prenatal or postnatal surgery.
Methods: The authors retrospectively identified patients from St. Louis Children's Hospital who were treated for MMC between 2016 and 2021. HC data were obtained from birth until the most recent follow-up or hydrocephalus treatment. Nonlinear least-squares regression analysis was performed to fit the data into four models: two-term power, Gompertz, West ontogenetic, and Weibull. Subsequently, the curves were assessed for their utility in predicting hydrocephalus treatment.
Results: Sixty-one patients (29 females [47.5%], 25 [41%] underwent prenatal surgery, mean gestational age at birth 36.6 weeks) were included in the study. The Weibull model best fit the HC data (prenatal adjusted R2 = 0.95, postnatal adjusted R2 = 0.95), while the Gompertz model had the worst fit (prenatal adjusted R2 = 0.56, postnatal adjusted R2 = 0.39) across both cohorts. Prenatal MMC repair patients had significantly larger HC measurements than their postnatal repair counterparts. The 50th percentile of the Weibull curve was determined as a useful threshold for hydrocephalus treatment: children with HC measurements that crossed and remained above this threshold were significantly more likely to have hydrocephalus treatment regardless of time of MMC repair (prenatal relative risk [RR] 10.0 [95% CI 1.424-70.220], sensitivity 85.7% [95% CI 0.499-0.984], and specificity 82.4% [95% CI 0.600-0.948]; postnatal RR 4.750 [95% CI 1.341-16.822], sensitivity 90.5% [95% CI 0.728-0.980], and specificity 75.0% [95% CI 0.471-0.924]). The HC growth curves of the MMC patients treated prenatally were significantly larger than the WHO HC curves (p < 0.001).
Conclusions: The Weibull model was identified as the HC growth curve with the best fit for MMC patients and serves as a useful predictor of hydrocephalus treatment. For MMC patients with hydrocephalus, prenatal repair patients fit the model well but postnatal repair patients did not, potentially indicating different mechanisms of hydrocephalus development. Those treated prenatally had significantly larger HC measurements compared with both the general population and those treated postnatally. Further study is needed to understand the long-term cognitive outcomes and optimal management of clinically asymptomatic patients with large HC measurements who were treated prenatally for MMC.