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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.

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

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