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赫尔曼斯基-普德拉克综合征

Hermansky-Pudlak syndrome.

作者信息

Krisp A, Hoffman R, Happle R, König A, Freyschmidt-Paul P

机构信息

Department of Dermatology, Philipp University, Deutschhausstr. 9, D-35033 Marburg, Germany.

出版信息

Eur J Dermatol. 2001 Jul-Aug;11(4):372-3.

Abstract

A 55-year-old man had oculocutaneous albinism and a history of frequent bruising following minimal trauma. The simultaneous occurrence of these features was first described by Hermansky and Pudlak in 1959. The Hermansky-Pudlak syndrome follows an autosomal recessive trait and is most frequently found in Puerto Rico and in the Swiss alps. It consists of the triad phenotype of hypopigmentation, prolonged bleeding time due to platelet storage pool deficiency and accumulation of ceroid pigment in lysosomal organelles. Other serious features are pulmonary fibrosis and granulomatous colitis. The disorder is caused by mutations in the HPS1 gene on chromosome 10q23. The HPS1 gene product is involved in the trafficking of melanosomes, platelet dense bodies, and lysosomes.

摘要

一名55岁男性患有眼皮肤白化病,并有轻微创伤后频繁出现瘀伤的病史。1959年,赫尔曼斯基和普德拉克首次描述了这些特征的同时出现。赫尔曼斯基-普德拉克综合征遵循常染色体隐性遗传特征,在波多黎各和瑞士阿尔卑斯山区最为常见。它由色素沉着不足、因血小板储存池缺乏导致的出血时间延长以及溶酶体细胞器中类蜡样色素积累这三种表型组成。其他严重特征包括肺纤维化和肉芽肿性结肠炎。该病症由位于10q23染色体上的HPS1基因突变引起。HPS1基因产物参与黑素小体、血小板致密体和溶酶体的运输。

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