罕见遗传性间质性肺病:图解文章。

IF 9 1区 医学 Q1 RESPIRATORY SYSTEM
European Respiratory Review Pub Date : 2024-11-13 Print Date: 2024-10-01 DOI:10.1183/16000617.0101-2024
Raphael Borie, Laureline Berteloot, Caroline Kannengiesser, Matthias Griese, Aurelie Cazes, Bruno Crestani, Alice Hadchouel, Marie Pierre Debray
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引用次数: 0

摘要

成人肺纤维化的主要单基因病因是端粒相关基因的突变。这些基因突变可能与肺外症状(肝病、血液病和皮肤病)有关,通常在放射学上表现为常见的间质性肺炎或无法分类的肺纤维化。在儿童中,肺纤维化的单基因病因主要是表面活性物质相关基因的突变。这些突变与肺外体征无关,通常在放射学上表现为不可分类的纤维化,并伴有囊肿,可导致成人胸壁畸形。本综述将讨论这些基因突变,以及大多数间质性肺病的单基因病因,包括干扰素相关基因、导致囊性肺病的基因突变、赫尔曼斯基-普德拉克综合征、肺泡蛋白病、溶血尿蛋白不耐受症和溶酶体贮积症,以及它们的肺部和肺外表现。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Rare genetic interstitial lung diseases: a pictorial essay.

The main monogenic causes of pulmonary fibrosis in adults are mutations in telomere-related genes. These mutations may be associated with extrapulmonary signs (hepatic, haematological and dermatological) and typically present radiologically as usual interstitial pneumonia or unclassifiable fibrosis. In children, the monogenic causes of pulmonary fibrosis are dominated by mutations in surfactant-related genes. These mutations are not associated with extrapulmonary signs and often manifest radiologically as unclassifiable fibrosis with cysts that can lead to chest wall deformities in adults. This review discusses these mutations, along with most of the monogenic causes of interstitial lung disease, including interferon-related genes, mutations in genes causing cystic lung disease, Hermansky-Pudlak syndrome, pulmonary alveolar proteinosis, lysinuric protein intolerance and lysosomal storage disorders, and their pulmonary and extrapulmonary manifestations.

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来源期刊
European Respiratory Review
European Respiratory Review Medicine-Pulmonary and Respiratory Medicine
CiteScore
14.40
自引率
1.30%
发文量
91
审稿时长
24 weeks
期刊介绍: The European Respiratory Review (ERR) is an open-access journal published by the European Respiratory Society (ERS), serving as a vital resource for respiratory professionals by delivering updates on medicine, science, and surgery in the field. ERR features state-of-the-art review articles, editorials, correspondence, and summaries of recent research findings and studies covering a wide range of topics including COPD, asthma, pulmonary hypertension, interstitial lung disease, lung cancer, tuberculosis, and pulmonary infections. Articles are published continuously and compiled into quarterly issues within a single annual volume.
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