波雷蒂-博尔特豪泽综合征的产前诊断--一例臼齿征象模拟病例报告

IF 2.7 3区 医学 Q3 NEUROSCIENCES Cerebellum Pub Date : 2024-12-01 Epub Date: 2024-08-12 DOI:10.1007/s12311-024-01728-4
Miguel Pereira-Macedo, Ana Grangeia, Ana Costa Braga, Ricardo Rolim, Alexandra Matias
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引用次数: 0

摘要

我们报告了一名 36 岁初产妇的波雷蒂-博尔特豪泽综合征(Poretti-Boltshauser Syndrome,PBS)产前诊断。在妊娠 22 周零 6 天时,胎儿超声检查发现小脑形状正常,但有高回声,所有幕上结构均正常。鉴别诊断包括海绵状血管瘤、毛细血管扩张症和小脑出血。随后的胎儿大脑核磁共振成像显示双侧小脑弥漫性改变、蚓部头尾直径缩小、小脑上梗病理性伸长和增厚,提示磨牙征。羊膜腔穿刺术和全外显子组测序确定了 LAMA1 基因的两个杂合截断变异:c.3099G > A (p.Trp1033Ter) 和 c.3699T > A (p.Tyr1233Ter),证实了 PBS 的存在。)确诊后,孕妇在妊娠 23 周零 5 天时终止妊娠。死后检查证实核磁共振成像结果与 PBS 一致。该病例强调了结合超声、核磁共振成像和遗传分析进行准确产前诊断的重要性,并强调了与 PBS 相关的分子多样性,包括存在磨牙征模拟和新型 c.3699T > A 变异。
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Prenatal Diagnosis of Poretti-Boltshauser Syndrome - a Case Report of a Molar Tooth Sign Mimic.

We report the prenatal diagnosis of Poretti-Boltshauser Syndrome (PBS) in a 36-year-old primigravida woman. At 22 weeks and 6 days of gestation, fetal ultrasound revealed a normally shaped but hyperechogenic cerebellum with all supratentorial structures appearing normal. Differential diagnosis included cavernous hemangioma, capillary telangiectasia, and cerebellar hemorrhage. Subsequent fetal cerebral MRI showed diffuse bilateral cerebellar modifications, reduced cranio-caudal diameter of the vermis, and pathological elongation and thickening of the superior cerebellar peduncles indicative of the molar tooth sign. Amniocentesis and whole exome sequencing identified two heterozygous truncating variants in the LAMA1 gene: c.3099G > A (p.Trp1033Ter) and c.3699T > A (p.Tyr1233Ter), confirming PBS.) Following the diagnosis, the pregnancy was terminated at 23 weeks and 5 days. Post-mortem examination supported the MRI findings consistent with PBS. This case highlights the importance of integrating ultrasound, MRI, and genetic analysis for accurate prenatal diagnosis and emphasizes the molecular diversity associated with PBS, including the presence of molar tooth sign mimics and a novel c.3699T > A variant.

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来源期刊
Cerebellum
Cerebellum 医学-神经科学
CiteScore
6.40
自引率
14.30%
发文量
150
审稿时长
4-8 weeks
期刊介绍: Official publication of the Society for Research on the Cerebellum devoted to genetics of cerebellar ataxias, role of cerebellum in motor control and cognitive function, and amid an ageing population, diseases associated with cerebellar dysfunction. The Cerebellum is a central source for the latest developments in fundamental neurosciences including molecular and cellular biology; behavioural neurosciences and neurochemistry; genetics; fundamental and clinical neurophysiology; neurology and neuropathology; cognition and neuroimaging. The Cerebellum benefits neuroscientists in molecular and cellular biology; neurophysiologists; researchers in neurotransmission; neurologists; radiologists; paediatricians; neuropsychologists; students of neurology and psychiatry and others.
期刊最新文献
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