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一名萨尔瓦多患者患成人起病型白质消失症伴卵巢功能衰竭

Adult-Onset White Matter Vanishing Disease With Ovarian Failure in a Salvadoran Patient.

作者信息

Flores Lazo Sara Loanny, Salazar-Orellana Jaime Leonardo I

机构信息

Department of Neurology, Instituto Salvadoreño del Seguro Social, San Salvador, SLV.

Department of Neurology and Psychiatry, Instituto Nacional de Ciencias Médicas y Nutrición Salvador Zubirán, Mexico City, MEX.

出版信息

Cureus. 2023 Jun 3;15(6):e39900. doi: 10.7759/cureus.39900. eCollection 2023 Jun.

DOI:10.7759/cureus.39900
PMID:37273681
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10239208/
Abstract

White matter vanishing disease is a type of leukodystrophy common in children with hypomyelination linked with ataxia, all of whom have an autosomal recessive inheritance. There are few reports of late-onset cases associated with ovarian failure. In Mesoamerican populations, this disease is mostly reported in children but rarely in adults. We present a case of a 35-year-old Salvadoran female patient with a history of menometrorrhagia, infertility, slowly progressive gait decline, paraparesis, spasticity, cerebellar ataxia, cognitive impairment with predominant executive dysfunction, learning difficulties, and emotional lability. A T2-weighted brain MRI and fluid-attenuated inversion recovery (FLAIR) images showed bilateral symmetrical areas of hyperintensities in the white matter with multiple foci of cystic degeneration within the hyperintense area. Two pathogenic variants were identified in the EIF2B5 gene. This case is of interest to increase awareness of late-onset leukodystrophies, widen the phenotypic spectrum of the disease, and constitute a valuable report for the international community, since it is a less frequently reported disease.

摘要

白质消失病是一种常见于儿童的脑白质营养不良症,与髓鞘形成不足相关,伴有共济失调,所有患者均为常染色体隐性遗传。关于与卵巢功能衰竭相关的迟发性病例报道较少。在中美洲人群中,这种疾病大多在儿童中报道,在成人中很少见。我们报告一例35岁的萨尔瓦多女性患者,有月经过多、不孕史,步态逐渐缓慢下降、双下肢轻瘫、痉挛、小脑共济失调、以执行功能障碍为主的认知障碍、学习困难和情绪不稳定。脑部T2加权磁共振成像(MRI)和液体衰减反转恢复(FLAIR)图像显示脑白质双侧对称高信号区,高信号区内有多个囊性变性灶。在EIF2B5基因中鉴定出两个致病变异。该病例有助于提高对迟发性脑白质营养不良症的认识,拓宽该疾病的表型谱,并为国际社会提供一份有价值的报告,因为这是一种报道较少的疾病。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/adc2/10239208/6ecf2c2d42f7/cureus-0015-00000039900-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/adc2/10239208/6ecf2c2d42f7/cureus-0015-00000039900-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/adc2/10239208/6ecf2c2d42f7/cureus-0015-00000039900-i01.jpg

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本文引用的文献

1
MRI Natural History of the Leukodystrophy Vanishing White Matter.MRI 脑白质消融性白质营养不良自然史
Radiology. 2021 Sep;300(3):671-680. doi: 10.1148/radiol.2021210110. Epub 2021 Jun 29.
2
Adult-onset vanishing white matter disease with the EIF2B2 gene mutation presenting as menometrorrhagia.成人发病进行性脑白质病伴 EIF2B2 基因突变以月经过多为表现。
BMC Neurol. 2019 Aug 22;19(1):203. doi: 10.1186/s12883-019-1429-9.
3
Natural History of Vanishing White Matter.脑白质消融症的自然病史。
Ann Neurol. 2018 Aug;84(2):274-288. doi: 10.1002/ana.25287. Epub 2018 Sep 6.
4
[Association between homozygous c.318A>GT mutation in exon 2 of the EIF2B5 gene and the infantile form of vanishing white matter leukoencephalopathy].[真核生物翻译起始因子2B亚基5(EIF2B5)基因第2外显子纯合c.318A>GT突变与婴儿型消失性白质脑病的关联]
Bol Med Hosp Infant Mex. 2017 Sep-Oct;74(5):364-369. doi: 10.1016/j.bmhimx.2017.07.002. Epub 2017 Sep 6.
5
Natural history of adult-onset eIF2B-related disorders: a multi-centric survey of 16 cases.成人起病的eIF2B相关疾病的自然史:16例多中心调查。
Brain. 2009 Aug;132(Pt 8):2161-9. doi: 10.1093/brain/awp171.
6
Vanishing white matter disease.脑白质消失症
Lancet Neurol. 2006 May;5(5):413-23. doi: 10.1016/S1474-4422(06)70440-9.
7
The large spectrum of eIF2B-related diseases.与真核起始因子2B(eIF2B)相关疾病的广泛谱系。
Biochem Soc Trans. 2006 Feb;34(Pt 1):22-9. doi: 10.1042/BST20060022.
8
Leukoencephalopathy with vanishing white matter: from magnetic resonance imaging pattern to five genes.伴脑白质消失的白质脑病:从磁共振成像模式到五个基因
J Child Neurol. 2003 Sep;18(9):639-45. doi: 10.1177/08830738030180091101.
9
Ovarian failure related to eukaryotic initiation factor 2B mutations.与真核生物起始因子2B突变相关的卵巢功能衰竭。
Am J Hum Genet. 2003 Jun;72(6):1544-50. doi: 10.1086/375404. Epub 2003 Apr 21.
10
Mutations in each of the five subunits of translation initiation factor eIF2B can cause leukoencephalopathy with vanishing white matter.翻译起始因子eIF2B的五个亚基中的每一个发生突变,都可能导致伴脑白质消失的白质脑病。
Ann Neurol. 2002 Feb;51(2):264-70. doi: 10.1002/ana.10112.