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孤立性蚓部发育不全的神经发育结局:一项单中心回顾性队列研究及文献综述

Neurodevelopmental Outcome in Isolated Vermian Hypoplasia: A Single-Center Retrospective Cohort Study and Literature Review.

作者信息

Cutillo Gianni, Zambon Marta, Faiola Stefano, Casati Daniela, Balestriero Marina, Doneda Chiara, Laoreti Arianna, Parazzini Cecilia, Spaccini Luigina, Lanna Mariano, Scelsa Barbara

机构信息

Department of Pediatric Neurology and Psychiatry, V. Buzzi Children's Hospital, ASST-FBF- Sacco, Via Castelvetro, 32, Milan, 20154, Italy.

Fetal Therapy Unit "U.Nicolini", V. Buzzi Children's Hospital, ASST-FBF-Sacco, Milan, Italy.

出版信息

Cerebellum. 2025 Mar 18;24(3):64. doi: 10.1007/s12311-025-01819-w.

DOI:10.1007/s12311-025-01819-w
PMID:40100516
Abstract

Vermian hypoplasia (VH) is characterized by reduced cerebellar vermis volume without significant hemispheric involvement. To date, data on long-term neurodevelopmental outcomes in isolated VH cases are limited, complicating prenatal counselling. This is single-center retrospective cohort study analyzing fetuses with posterior fossa abnormalities (PFA) diagnosed between 2010 and 2021. VH cases were confirmed through ultrasonography and fetal MRI, excluding non-isolated anomalies or ischemic/hemorrhagic etiologies. We collected data on Neurodevelopmental outcomes through chart review and structured interviews with the caregiver. We also provided a literature review summarizing prior research on VH outcomes. Out of 45 cases with (PFA), isolated VH was found in 15 children and was confirmed in 7 cases through fetal MRI. Among these patients, median gestational age at diagnosis was 22.5 weeks, with follow-ups extending to a median age of 6 years. Age-appropriate neurodevelopmental outcomes were normal or showed minimal deficits in 6/7 (70%). Literature review highlighted a wide outcome variability, partly due to varying diagnostic criteria and follow-up protocols. Long-term prognosis is influenced by various factors, including genetic testing and environmental support. In isolated VH, outcomes are generally favorable, though mild learning or motor coordination deficits may emerge. Broader normative biometrics and consistent neurodevelopmental evaluations are critical for improved prognostic precision. VH presents diagnostic and prognostic challenges due to variability in outcomes and limited longitudinal data. A multidisciplinary approach, including prenatal imaging, genetic testing, and structured follow-up, is essential for effective family counseling and developmental support. Prospective studies with larger cohorts are warranted to establish clearer guidelines.

摘要

蚓部发育不全(VH)的特征是小脑蚓部体积减小,而半球无明显受累。迄今为止,关于孤立性VH病例长期神经发育结局的数据有限,这使得产前咨询变得复杂。这是一项单中心回顾性队列研究,分析了2010年至2021年期间诊断为后颅窝异常(PFA)的胎儿。VH病例通过超声检查和胎儿MRI确诊,排除非孤立性异常或缺血/出血性病因。我们通过病历审查和与照顾者的结构化访谈收集了神经发育结局的数据。我们还提供了一篇文献综述,总结了先前关于VH结局的研究。在45例PFA病例中,15名儿童被发现为孤立性VH,其中7例通过胎儿MRI确诊。在这些患者中,诊断时的中位孕周为22.5周,随访至中位年龄6岁。6/7(70%)的患者年龄适宜的神经发育结局正常或仅有轻微缺陷。文献综述强调了结局的广泛变异性,部分原因是诊断标准和随访方案不同。长期预后受多种因素影响,包括基因检测和环境支持。在孤立性VH中,结局通常较好,尽管可能会出现轻度学习或运动协调缺陷。更广泛的标准生物测量和一致的神经发育评估对于提高预后准确性至关重要。由于结局的变异性和纵向数据有限,VH带来了诊断和预后挑战。多学科方法,包括产前影像学、基因检测和结构化随访,对于有效的家庭咨询和发育支持至关重要。有必要进行更大队列的前瞻性研究以建立更明确的指南。

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本文引用的文献

1
Relative prevalence and outcome of fetal posterior fossa abnormality.胎儿后颅窝异常的相对患病率及结局
J Paediatr Child Health. 2023 Jan;59(1):107-115. doi: 10.1111/jpc.16254. Epub 2022 Nov 1.
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Refining the Neuroimaging Definition of the Dandy-Walker Phenotype.细化 Dandy-Walker 表型的神经影像学定义。
AJNR Am J Neuroradiol. 2022 Oct;43(10):1488-1493. doi: 10.3174/ajnr.A7659. Epub 2022 Sep 22.
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Short- and Long-Term Outcomes of Prenatally Diagnosed Dandy-Walker Malformation, Vermian Hypoplasia, and Blake Pouch Cyst.
产前诊断的Dandy-Walker畸形、小脑蚓部发育不全和Blake囊肿的短期和长期预后
J Child Neurol. 2021 Oct;36(12):1111-1119. doi: 10.1177/08830738211049115. Epub 2021 Nov 10.
4
ISUOG Practice Guidelines (updated): sonographic examination of the fetal central nervous system. Part 2: performance of targeted neurosonography.国际妇产科超声学会实践指南(更新版):胎儿中枢神经系统超声检查。第2部分:针对性神经超声检查的操作
Ultrasound Obstet Gynecol. 2021 Apr;57(4):661-671. doi: 10.1002/uog.23616. Epub 2021 Mar 18.
5
Diagnostic Approach to Cerebellar Hypoplasia.小脑发育不良的诊断方法。
Cerebellum. 2021 Aug;20(4):631-658. doi: 10.1007/s12311-020-01224-5. Epub 2021 Feb 3.
6
Developmental outcomes in children with congenital cerebellar malformations.先天性小脑畸形患儿的发育结局。
Dev Med Child Neurol. 2019 Mar;61(3):350-358. doi: 10.1111/dmcn.14059. Epub 2018 Oct 15.
7
Enlarged posterior fossa on prenatal imaging: differential diagnosis, associated anomalies and postnatal outcome.产前影像学检查发现的后颅窝扩大:鉴别诊断、相关异常及产后结局
Acta Obstet Gynecol Scand. 2017 Jul;96(7):837-843. doi: 10.1111/aogs.13131. Epub 2017 Apr 10.
8
Systematic review and meta-analysis of isolated posterior fossa malformations on prenatal imaging (part 2): neurodevelopmental outcome.产前影像学孤立性后颅窝畸形的系统评价和荟萃分析(第2部分):神经发育结局
Ultrasound Obstet Gynecol. 2016 Jul;48(1):28-37. doi: 10.1002/uog.15755.
9
Pre- and Postnatal Neuroimaging of Congenital Cerebellar Abnormalities.先天性小脑异常的产前和产后神经影像学检查
Cerebellum. 2016 Feb;15(1):5-9. doi: 10.1007/s12311-015-0699-z.
10
Systematic review and meta-analysis of isolated posterior fossa malformations on prenatal ultrasound imaging (part 1): nomenclature, diagnostic accuracy and associated anomalies.系统评价和荟萃分析产前超声影像学孤立性后颅窝畸形(第 1 部分):命名、诊断准确性和相关异常。
Ultrasound Obstet Gynecol. 2016 Jun;47(6):690-7. doi: 10.1002/uog.14900. Epub 2016 May 10.