Suppr超能文献

用兔乳中重组人α-葡萄糖苷酶对庞贝氏病进行酶疗法。

Enzyme therapy for pompe disease with recombinant human alpha-glucosidase from rabbit milk.

作者信息

Van den Hout J M, Reuser A J, de Klerk J B, Arts W F, Smeitink J A, Van der Ploeg A T

机构信息

Department of Pediatrics, Sophia Children's Hospital, University Hospital Rotterdam, The Netherlands.

出版信息

J Inherit Metab Dis. 2001 Apr;24(2):266-74. doi: 10.1023/a:1010383421286.

Abstract

Pompe disease is a metabolic myopathy caused by deficiency of lysosomal acid alpha-glucosidase. In this report we review the first 36 weeks of a clinical study on the safety and efficacy of enzyme therapy aimed at correcting the deficiency. Four patients with infantile Pompe disease were enrolled. They received recombinant human alpha-glucosidase from transgenic rabbit milk. The product is generally well tolerated and reaches the primary target tissues. Normalization of alpha-glucosidase activity in skeletal muscle was obtained and degradation of PAS-positive material was seen in tissue sections. The clinical condition of all patients improved. The effect on heart was most significant, with an impressive reduction of the left ventricular mass index (LVMI). Motor function improved. The positive preliminary results stimulate continuation and extension of efforts towards the realization of enzyme therapy for Pompe disease.

摘要

庞贝氏病是一种由溶酶体酸性α-葡萄糖苷酶缺乏引起的代谢性肌病。在本报告中,我们回顾了一项旨在纠正该缺乏症的酶疗法安全性和有效性的临床研究的前36周。招募了4名婴儿型庞贝氏病患者。他们接受了来自转基因兔奶的重组人α-葡萄糖苷酶。该产品总体耐受性良好,并能到达主要靶组织。骨骼肌中α-葡萄糖苷酶活性恢复正常,并且在组织切片中可见PAS阳性物质降解。所有患者的临床状况均有所改善。对心脏的影响最为显著,左心室质量指数(LVMI)显著降低。运动功能得到改善。这些积极的初步结果促使人们继续并扩大努力,以实现庞贝氏病的酶疗法。

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验