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一例伴有呼吸困难的赫尔曼斯基-普德拉克综合征病例。

A case of Hermansky-Pudlak with dyspnea.

作者信息

Takaldani Ali Hossein Samadi, Javanshir Nima, Salimi Maryam, Negaresh Mohammad

机构信息

Department of Internal Medicine (Pulmonology Division), School of Medicine, Ardabil University of Medical Sciences, Ardabil, Iran.

Faculty of Medicine, School of Medicine, Ardabil University of Medical Sciences, Ardabil, Iran.

出版信息

Oxf Med Case Reports. 2023 Feb 27;2023(2):omad001. doi: 10.1093/omcr/omad001. eCollection 2023 Feb.

Abstract

Hermansky-Pudlak syndrome (HPS) is a rare multisystem disorder inherited in an autosomal recessive manner. Its prevalence is 1 in 500 000 to 1 000 000 people worldwide. The cause of this disorder is genetic mutations that lead to defective organelles of lysosomes. In this report, a 49-year-old man is introduced who was referred to the medical center with ocular albinism and recently exacerbated shortness of breath. Imaging showed peripheral reticular opacities, ground-glass opacities of the lungs with subpleural sparing in some regions, and thickening of bronchovascular bundles, which were all in favor of non-specific interstitial pneumonia. This imaging pattern is an unusual finding in a patient with HPS.

摘要

Hermansky-Pudlak综合征(HPS)是一种罕见的以常染色体隐性方式遗传的多系统疾病。其在全球的患病率为五十万分之一至一百万分之一。这种疾病的病因是导致溶酶体细胞器缺陷的基因突变。在本报告中,介绍了一名49岁男性,他因眼部白化病被转诊至医疗中心,近期出现气短加重。影像学检查显示外周网状阴影、肺部磨玻璃样阴影,部分区域胸膜下未受累,以及支气管血管束增粗,这些均提示非特异性间质性肺炎。这种影像学表现在HPS患者中是不常见的发现。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0721/9969820/abac07072c47/omad001f1.jpg

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