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先天性弓形虫病继发脑积水的结局

Outcomes of hydrocephalus secondary to congenital toxoplasmosis.

作者信息

McLone David, Frim David, Penn Richard, Swisher Charles N, Heydemann Peter, Boyer Kenneth M, Noble A Gwendolyn, Rabiah Peter K, Withers Shawn, Wroblewski Kristen, Karrison Theodore, Hutson Samuel, Wheeler Kelsey, Cohen William, Lykins Joseph, McLeod Rima

机构信息

1Northwestern University and Lurie Children's Hospital and Medical Center, Chicago.

2Section of Neurosurgery.

出版信息

J Neurosurg Pediatr. 2019 Sep 6;24(5):601-608. doi: 10.3171/2019.6.PEDS18684. Print 2019 Nov 1.

DOI:10.3171/2019.6.PEDS18684
PMID:31491752
Abstract

OBJECTIVE

Hydrocephalus occurs in children with congenital toxoplasmosis and can lead to severe disability. In these cases, the decision to intervene is often influenced by the expectation of neurological recovery. In this study, clinical responses to neurosurgical intervention in children with hydrocephalus secondary to congenital toxoplasmosis are characterized.

METHODS

Sixty-five participants with hydrocephalus due to congenital Toxoplasma gondii infection were evaluated as part of the National Collaborative Chicago-based Congenital Toxoplasmosis Study, and their neuroradiographic findings were reviewed. Clinical outcomes were scored on the basis of cognition and motor skills through the use of IQ scores and Gross Motor Function Classification System (GMFCS) level. Outcomes were then analyzed in relation to approach to management, anatomy of hydrocephalus, and time from diagnosis of hydrocephalus to surgical intervention.

RESULTS

There was considerable variation in the outcomes of patients whose hydrocephalus was treated in early life, ranging from normal cognitive and motor function to profound developmental delay and functional limitation. Of the 65 participants included in the study, IQ and GMFCS level were available for 46 (70.8%). IQ and motor score were highly correlated (r = -0.82, p < 0.001). There were people with differing patterns of hydrocephalus or thickness of cortical mantle on initial presentation who had favorable outcomes. Time to neurosurgical intervention data were available for 31 patients who underwent ventriculoperitoneal (VP) shunt placement. Delayed shunt placement beyond 25 days after diagnosis of hydrocephalus was associated with greater cognitive impairment (p = 0.02). Motor impairment also appeared to be associated with shunt placement beyond 25 days but the difference did not achieve statistical significance (p = 0.13). Among those with shunt placement within 25 days after diagnosis (n = 19), the mean GMFCS level was 1.9 ± 1.6 (range 1-5). Five (29.4%) of 17 of these patients were too disabled to participate in formal cognitive testing, after excluding 2 patients with visual difficulties or language barriers that precluded IQ testing. Of the patients who had VP shunt placement 25 or more days after diagnosis (n = 12), the mean GMFCS level was 2.7 ± 1.4 (range 1-4). Of these, 1 could not participate in IQ testing due to severe visual difficulties and 8 (72.7%) of the remaining 11 due to cognitive disability.

CONCLUSIONS

VP shunt placement in patients with hydrocephalus caused by congenital toxoplasmosis can contribute to favorable clinical outcomes, even in cases with severe hydrocephalus on neuroimaging. Shunt placement within 25 days of diagnosis was statistically associated with more favorable cognitive outcomes. Motor function appeared to follow the same pattern although it did not achieve statistical significance.

摘要

目的

先天性弓形虫病患儿会发生脑积水,并可能导致严重残疾。在这些病例中,干预决策往往受到神经功能恢复预期的影响。本研究对先天性弓形虫病继发脑积水患儿神经外科干预的临床反应进行了特征描述。

方法

作为基于芝加哥的全国先天性弓形虫病协作研究的一部分,对65例因先天性弓形虫感染导致脑积水的参与者进行了评估,并对他们的神经影像学检查结果进行了回顾。通过使用智商分数和粗大运动功能分类系统(GMFCS)水平,根据认知和运动技能对临床结果进行评分。然后分析结果与治疗方法、脑积水解剖结构以及从脑积水诊断到手术干预的时间之间的关系。

结果

早年接受脑积水治疗的患者结果差异很大,从正常的认知和运动功能到严重的发育迟缓及功能受限。在纳入研究的65名参与者中,46名(70.8%)有智商和GMFCS水平数据。智商与运动评分高度相关(r = -0.82,p < 0.001)。初始表现时脑积水模式或皮质层厚度不同的患者也有良好的结果。3个1例接受脑室腹腔(VP)分流置管的患者有从诊断到神经外科干预的时间数据。脑积水诊断后超过25天延迟进行分流置管与更严重的认知障碍相关(p = 0.02)。运动障碍似乎也与超过25天进行分流置管有关,但差异未达到统计学意义(p = 0.13)。在诊断后25天内进行分流置管的患者中(n = 19),GMFCS平均水平为1.9 ± 1.6(范围1 - 5)。在排除2例因视力困难或语言障碍而无法进行智商测试的患者后,这些患者中有5例(29.4%)的17例因残疾严重而无法参加正式认知测试。在诊断后25天或更长时间进行VP分流置管的患者中(n = 12),GMFCS平均水平为2.7 ± 1.4(范围1 - 4)。其中,1例因严重视力困难无法参加智商测试,其余11例中有8例(72.7%)因认知残疾无法参加。

结论

先天性弓形虫病所致脑积水患者进行VP分流置管可带来良好的临床结果,即使在神经影像学显示严重脑积水的病例中也是如此。诊断后25天内进行分流置管在统计学上与更有利的认知结果相关。运动功能似乎遵循相同模式,尽管未达到统计学意义。

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