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与转甲状腺素蛋白基因Val30Met相关的大量软脑膜淀粉样变性

Massive leptomeningeal amyloidosis associated with a Val30Met transthyretin gene.

作者信息

Herrick M K, DeBruyne K, Horoupian D S, Skare J, Vanefsky M A, Ong T

机构信息

Department of Pathology, Santa Clara Valley Medical Center, San Jose, CA 95128-2699, USA.

出版信息

Neurology. 1996 Oct;47(4):988-92. doi: 10.1212/wnl.47.4.988.

Abstract

We report a 69-year-old woman of Mexican origin with a 6-year history of progressive paresis, mild peripheral neuropathy, and recent onset of fluctuating mental status. Head and spinal MRI revealed contrast enhancing thickened meninges which on biopsy disclosed amyloid deposition. Immunohistochemistry identified the amyloid as transthyretin (TTR), and polymerase chain reaction/restriction fragment length polymorphism analysis of blood revealed a Val30Met mutation in one of her TTR genes. This mutation causes familial (hereditary) amyloidotic polyneuropathy of the Portuguese type (FAP 1). However, unlike FAP 1, in which peripheral neuropathy is a dominant feature, our patient's clinical manifestations, which included communicating hydrocephalus and myelopathy, were more suggestive of familial oculoleptomeningeal amyloidosis (FOLMA). In summary, the clinical presentation of TTR Met 30 mutation is more varied than previously suspected, and leptomeningeal amyloidosis should be considered in the differential diagnosis of obscure conditions involving meninges.

摘要

我们报告了一位69岁的墨西哥裔女性,她有6年进行性轻瘫病史、轻度周围神经病变,近期出现精神状态波动。头部和脊髓磁共振成像(MRI)显示脑膜增厚并强化,活检发现有淀粉样蛋白沉积。免疫组化鉴定该淀粉样蛋白为转甲状腺素蛋白(TTR),血液聚合酶链反应/限制性片段长度多态性分析显示其一个TTR基因存在Val30Met突变。此突变导致葡萄牙型家族性(遗传性)淀粉样多神经病(FAP 1)。然而,与以周围神经病变为主要特征的FAP 1不同,我们患者的临床表现包括交通性脑积水和脊髓病,更符合家族性眼软脑膜淀粉样变性(FOLMA)。总之,TTR Met 30突变的临床表现比之前怀疑的更多样化,在涉及脑膜的疑难病症鉴别诊断中应考虑软脑膜淀粉样变性。

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