Kato Takafumi, Akagawa Shinobu, Kusaka Kei, Kawashima Masahiro, Ohshima Nobuharu, Kitani Masashi, Hebisawa Akira, Matsui Hirotoshi
Center for Pulmonary Diseases, National Hospital Organization Tokyo National Hospital, Tokyo, Japan.
Division of Pathology, National Hospital Organization Tokyo National Hospital, Tokyo, Japan.
Respir Med Case Rep. 2020 Dec 31;32:101332. doi: 10.1016/j.rmcr.2020.101332. eCollection 2021.
Yellow nail syndrome (YNS) is a rare entity characterized by thickened yellowish nails, lymphedema and respiratory manifestations such as pleural effusion. Lymphatic dysfunction is considered as a cause of YNS. However, evidence of systemic dilatation/hyperplasia of lymphatics based on autopsy in YNS is not available. In this report, autopsy revealed dilatation and hyperplasia of lymphatic vessels in lungs, visceral and parietal pleurae, and intestines. We identified the direct opening of lymphatic vessels of the visceral pleura to the pleural cavity, which indicated the pathophysiology of uncontrollable pleural effusion in YNS. The current case was compromised with primary biliary cholangitis (PBC). The onset of PBC seemed to be related with the progression of YNS.
黄甲综合征(YNS)是一种罕见的病症,其特征为指甲增厚发黄、淋巴水肿以及诸如胸腔积液等呼吸系统表现。淋巴功能障碍被认为是YNS的一个病因。然而,基于YNS尸检的淋巴管系统性扩张/增生的证据并不存在。在本报告中,尸检显示肺、脏层和壁层胸膜以及肠道的淋巴管扩张和增生。我们发现脏层胸膜的淋巴管直接通向胸腔,这表明了YNS中胸腔积液难以控制的病理生理学机制。本例患者合并原发性胆汁性胆管炎(PBC)。PBC的发病似乎与YNS的进展有关。