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异染性脑白质营养不良的眼部表现。不同临床和基因变异型的电子显微镜及酶学研究。

Ocular findings in metachromatic leukodystrophy. An electron microscopic and enzyme study in different clinical and genetic variants.

作者信息

Libert J, Van Hoof F, Toussaint D, Roozitalab H, Kenyon K R, Green W R

出版信息

Arch Ophthalmol. 1979 Aug;97(8):1495-504. doi: 10.1001/archopht.1979.01020020157015.

Abstract

Histopathological studies of the eyes from three patients affected with the infantile form of metachromatic leukodystrophy (MLD) showed the storage of metachromatic complex lipids in the retinal ganglion cells, in the optic nerve and the ciliary nerves, as well as the storage of a mucopolysaccharide-like material in the nonpigmented epithelium of the ciliary body. The lesions were limited to the optic, ciliary, and sensory nerves in a fourth patient with the juvenile form of the disorder. These morphological aspects, which are probably related to differences in sulfatase A activities, may explain the variability of the ocular manifestations in metachromatic leukodystrophy. Seven children affected with infantile MLD or with mucosulfatidosis were examined by conjunctival biopsy. Typical lesions of the sensory nerves were obvious and allowed the diagnosis of the disease. However, it seemed impossible to separate the different forms by histopathological studies only. The tear enzymes were assayed in most of the cases and demonstrated a profound deficiency of arylsulfatase A, or of arylsulfatase A and B, in the classical MLD and in mucosulfatidosis, respectively.

摘要

对三名患有婴儿型异染性脑白质营养不良(MLD)患者的眼部进行组织病理学研究显示,异染性复合脂质在视网膜神经节细胞、视神经和睫状神经中蓄积,同时一种黏多糖样物质在睫状体的无色素上皮中蓄积。在第四名患有青少年型该疾病的患者中,病变仅限于视神经、睫状神经和感觉神经。这些形态学特征可能与芳基硫酸酯酶A活性的差异有关,这或许可以解释异染性脑白质营养不良眼部表现的变异性。对七名患有婴儿型MLD或黏脂贮积症的儿童进行了结膜活检。感觉神经的典型病变很明显,从而得以诊断该疾病。然而,似乎仅通过组织病理学研究无法区分不同类型。在大多数病例中对泪液酶进行了检测,结果表明在经典型MLD和黏脂贮积症中,分别存在芳基硫酸酯酶A或芳基硫酸酯酶A和B的严重缺乏。

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