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一名具有异常临床特征女孩的β-葡萄糖醛酸酶缺乏症

Beta-glucuronidase deficiency in a girl with unusual clinical features.

作者信息

Pfeiffer R A, Kresse H, Bäumer N, Sattinger E

出版信息

Eur J Pediatr. 1977 Oct 12;126(3):155-61. doi: 10.1007/BF00442197.

Abstract

beta-glucuronidase deficiency in fibroblasts, leucocytes and in serum and increased urinary excretion of mucopolysaccharides were found in a girl, now 13 years old, who exhibits some features of a mucopolysaccharidosis such as moderate mental deficiency, craniofacial dysmorphism, a short neck, protruding sternum, vertebral deformities and corneal clouding. Coarse granulations were found in her leucocytes. The liver and spleen are not enlarged and there is no gingival hyperplasia. Additional features, hitherto undescribed, are hydronephrosis and defective ossification of the medial carpal and tarsal bones. Low enzyme activity in the parents and a normal brother suggests heterozygosity.

摘要

在一名现年13岁的女孩身上发现其成纤维细胞、白细胞及血清中β-葡萄糖醛酸酶缺乏,且尿中黏多糖排泄增加。该女孩表现出一些黏多糖贮积症的特征,如中度智力缺陷、颅面畸形、短颈、胸骨突出、脊柱畸形和角膜混浊。在她的白细胞中发现了粗大颗粒。肝脏和脾脏未肿大,也没有牙龈增生。其他迄今未描述的特征是肾积水以及腕骨和跗骨内侧骨化缺陷。父母及一个正常兄弟的酶活性较低,提示为杂合子状态。

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