Atypical STING-Vasculopathy Phenotype: Definite Usual Interstitial Pneumonia (UIP)-Pattern CT With Mixed Fibrotic Histology: A Case Report

非典型STING血管病表型:典型的寻常型间质性肺炎(UIP)CT表现伴混合纤维化组织学:病例报告

阅读:1

Abstract

STING‑associated vasculopathy with onset in infancy (SAVI) is a rare autoinflammatory disorder that causes systemic inflammation, vasculopathy, and progressive interstitial lung disease (ILD). The pulmonary manifestations of SAVI typically resemble a nonspecific interstitial pneumonia (NSIP) pattern both radiologically and histologically. We present a case of a 20‑year‑old male with genetically confirmed SAVI who developed acute hypoxic respiratory failure triggered by respiratory syncytial virus infection, despite appropriate treatment. Imaging revealed extensive subpleural fibrosis, traction bronchiectasis, and honeycombing. These findings were observed in the setting of a CT appearance strongly consistent with a definite usual interstitial pneumonia (UIP) pattern. Ultimately, the patient underwent a lung transplant, and histopathology of the explanted lungs revealed fibrotic changes, including diffuse interstitial thickening, lymphoid aggregates, and honeycombing, without definitive fibroblastic foci, indicating a mixed UIP and NSIP pattern of fibrosis. This case illustrates a rare UIP‑pattern imaging phenotype in SAVI‑associated ILD, despite a mixed UIP‑NSIP histologic pattern of pulmonary fibrosis, and highlights the potential for acute viral infection to precipitate decompensation in patients with interferon‑driven pulmonary fibrosis. Written informed consent was obtained from the patient for publication of this report and accompanying images.

特别声明

1、本页面内容包含部分的内容是基于公开信息的合理引用;引用内容仅为补充信息,不代表本站立场。

2、若认为本页面引用内容涉及侵权,请及时与本站联系,我们将第一时间处理。

3、其他媒体/个人如需使用本页面原创内容,需注明“来源:[生知库]”并获得授权;使用引用内容的,需自行联系原作者获得许可。

4、投稿及合作请联系:info@biocloudy.com。