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桑菲利波综合征(MPS III)管理的当前概念:叙述性综述

Current Concepts in the Management of Sanfilippo Syndrome (MPS III): A Narrative Review.

作者信息

Alyazidi Anas S, Muthaffar Osama Y, Baaishrah Layan S, Shawli Mohammed K, Jambi Abdulaziz T, Aljezani Maram A, Almaghrabi Majdah A

机构信息

Pediatrics, King Abdulaziz University Faculty of Medicine, Jeddah, SAU.

Faculty of Pharmacy, King Abdulaziz University Hospital, Jeddah, SAU.

出版信息

Cureus. 2024 Apr 11;16(4):e58023. doi: 10.7759/cureus.58023. eCollection 2024 Apr.

Abstract

Sanfilippo syndrome is a childhood-onset (1-4 years) autosomal recessive lysosomal storage disease that presents as a neurodegenerative disease by targeting the brain and spinal cord. It is also known as mucopolysaccharidosis III. Mucopolysaccharidosis III is divided into four subtypes (A, B, C, or D). It can cause delayed speech, behavior problems, and features of autism spectrum disorder. Sanfilippo syndrome is of a higher prevalence within consanguineous families that carry its gene alteration. If both parents have a nonfunctional copy of a gene linked to this condition, their children will have a 25% (1 in 4) chance of developing the disease. In Saudi Arabia, the incidence rate is estimated at 2 per 100,000 live births. Recent research focused on promising treatment approaches, such as gene therapy, modified enzyme replacement therapy, and stem cells. These approaches work by exogenous administration of the proper version of the mutant enzyme (enzyme replacement therapy), cleaning the defective enzyme in individuals with glycolipid storage disorders (substrate reduction therapy), or using a pharmacological chaperone to target improperly folded proteins. However, there is currently no approved curative medication for Sanfilippo syndrome that can effectively halt or reverse the disorder.

摘要

桑菲利波综合征是一种儿童期发病(1至4岁)的常染色体隐性溶酶体贮积病,通过侵袭大脑和脊髓而表现为神经退行性疾病。它也被称为黏多糖贮积症III型。黏多糖贮积症III型分为四个亚型(A、B、C或D)。它可导致言语发育迟缓、行为问题以及自闭症谱系障碍的特征。桑菲利波综合征在携带其基因改变的近亲家庭中患病率较高。如果父母双方都有与这种疾病相关的无功能基因拷贝,他们的孩子患该病的几率为25%(四分之一)。在沙特阿拉伯,发病率估计为每10万例活产中有2例。最近的研究集中在有前景的治疗方法上,如基因治疗、改良酶替代疗法和干细胞。这些方法的作用方式包括外源性给予突变酶的正确版本(酶替代疗法)、清除患有糖脂贮积症个体中的缺陷酶(底物减少疗法),或使用药理学伴侣靶向折叠不当的蛋白质。然而,目前尚无获批的可有效阻止或逆转桑菲利波综合征的治愈性药物。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/54e8/11087936/700e2dfe61b7/cureus-0016-00000058023-i01.jpg

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