Suppr超能文献

克拉伯病:伴有多发性硬化症家族史的严重新生儿表现。

Krabbe disease: severe neonatal presentation with a family history of multiple sclerosis.

作者信息

Sahai Inderneel, Baris Hagit, Kimonis Virginia, Levy Harvey L

机构信息

Division of Genetics and metabolism, Harvard Medical School and Children's Hospital, Boston, MA, USA.

出版信息

J Child Neurol. 2005 Oct;20(10):826-8. doi: 10.1177/08830738050200100901.

Abstract

Krabbe disease, also known as globoid cell leukodystrophy, is a rare autosomal recessive disorder caused by a deficiency of a lysosomal enzyme, galactocerebrosidase. This defect prevents normal turnover of the galactolipids and results in progressive demyelination. In the infantile form, symptoms typically present at 3 to 6 months of age with subsequent neurologic deterioration. We report a case with presentation on day 7 of life and rapid progression culminating in death at 10 weeks. Galactocerebrosidase activity was absent in the leukocytes, and a 30 kb deletion in the GALC gene was found. To our knowledge, this is the earliest reported death from Krabbe disease. Several family members have multiple sclerosis, which is also a demyelinating disorder. We propose that the neonatal expression could be an example of complementary gene interaction in which coinheritance of a predisposition to multiple sclerosis led to the unusual early manifestation and rapid course of Krabbe disease in this infant.

摘要

克拉伯病,也称为球形细胞脑白质营养不良,是一种罕见的常染色体隐性疾病,由溶酶体酶半乳糖脑苷脂酶缺乏引起。这种缺陷阻止了半乳糖脂的正常更新,导致进行性脱髓鞘。在婴儿型中,症状通常在3至6个月大时出现,随后出现神经功能恶化。我们报告了一例在出生第7天出现症状并迅速进展,最终在10周时死亡的病例。白细胞中缺乏半乳糖脑苷脂酶活性,并且在GALC基因中发现了一个30 kb的缺失。据我们所知,这是克拉伯病最早报道的死亡病例。几个家庭成员患有多发性硬化症,这也是一种脱髓鞘疾病。我们认为,新生儿期发病可能是互补基因相互作用的一个例子,即多发性硬化症易感性的共同遗传导致了该婴儿克拉伯病异常的早期表现和快速病程。

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验