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一例土耳其的半乳糖唾液酸贮积症患者,其CTSA基因存在新的纯合突变。

A Turkish case of galactosialidosis with a new homozygous mutation in CTSA gene.

作者信息

Kartal Ayşe, Aydın Kürşad

机构信息

Division of Child Neurology, Selçuk University Faculty of Medicine, Alaeddin Kampusü, Selçuklu, Konya, Türkiye.

Division of Child Neurology, Gazi University Faculty of Medicine, Ankara, Turkey.

出版信息

Metab Brain Dis. 2017 Aug;32(4):973-975. doi: 10.1007/s11011-017-0042-0. Epub 2017 May 30.

Abstract

Galactosialidosis is an autosamal reressive lysosomal storage disease caused by a combined deficiency of lysosomal β-galactosidase and neuraminidase, due to a primary defect in protective protein/cathepsin A. Three subtypes are recognized: the early infantile type, the late infantile type, and the juvenile/adult type. We report here a female patient with early infantile galactosialidosis who was born at 35 weeks of gestation. After birth she remained at the neonatal intensive care unit. Physical examination revealed, coarse facial features, hepatomegaly, cardiac murmur and diffuse hypotonia. The patient's mother had a past history of fetal hydrops history. The diagnosis of galactosialidosis was confirmed by decreased activity of β-galactosidase and undetectable neuraminidase activity in fibroblasts. Genetic examination revealed a new homozygous mutation (c.1284delG) in the CTSA gene.

摘要

半乳糖唾液酸贮积症是一种常染色体隐性溶酶体贮积病,由溶酶体β-半乳糖苷酶和神经氨酸酶联合缺乏引起,原因是保护蛋白/组织蛋白酶A存在原发性缺陷。已确认有三种亚型:早发型婴儿型、晚发型婴儿型和青少年/成人型。我们在此报告一名孕35周出生的早发型婴儿型半乳糖唾液酸贮积症女性患者。出生后她一直在新生儿重症监护室。体格检查发现面部特征粗糙、肝肿大、心脏杂音和弥漫性肌张力减退。患者的母亲有胎儿水肿病史。通过成纤维细胞中β-半乳糖苷酶活性降低和神经氨酸酶活性检测不到,确诊为半乳糖唾液酸贮积症。基因检查发现CTSA基因有一个新的纯合突变(c.1284delG)。

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