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胎儿期的桑菲利波综合征A型

Sanfilippo A syndrome in the fetus.

作者信息

Greenwood R S, Hillman R E, Alcala H, Sly W S

出版信息

Clin Genet. 1978 Mar;13(3):241-50. doi: 10.1111/j.1399-0004.1978.tb01177.x.

Abstract

A family is reported in which Sanfilippo A syndrome affected three siblings: the proband and twin premature infants. The feasibility of intrauterine diagnosis of mucopolysaccharidoses (MPS) Type IIIA, was demonstrated by the excessive accumulation of 35SO4-mucopolysaccharides in fibroblasts cultured from amniotic fluid obtained by amniocentesis Cross-correction studies and enzymatic analysis of cultured skin fibroblasts from the proband and the infants revealed the absence of the MPS IIIA correction factor, heparan sulfate sulfatase. However, when the premature infants expired shortly after birth, no central nervous system histopathology or ultrastructural abnormalities were found. From these observations it would appear the the third trimester fetus with MPS type IIIA has little CNS involvement.

摘要

据报道,一个家庭中有三名兄弟姐妹患有Sanfilippo A综合征:先证者和一对早产双胞胎。通过羊膜穿刺术获取羊水培养的成纤维细胞中35SO4 - 粘多糖过度积累,证明了宫内诊断IIIA型粘多糖贮积症(MPS)的可行性。对先证者和婴儿培养的皮肤成纤维细胞进行交叉校正研究和酶分析,发现缺乏MPS IIIA校正因子硫酸乙酰肝素硫酸酯酶。然而,早产婴儿出生后不久死亡,未发现中枢神经系统组织病理学或超微结构异常。从这些观察结果来看,孕晚期患有IIIA型MPS的胎儿中枢神经系统受累程度似乎很小。

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