Cong Cung-Van, Anh Tran-Thi Tuan, Ly Tran-Thi, Duc Nguyen Minh
Department of Radiology, National Lung Hospital, Ha Noi, Vietnam.
Department of Pathology, National Lung Hospital, Ha Noi, Vietnam.
Radiol Case Rep. 2022 Mar 21;17(5):1646-1655. doi: 10.1016/j.radcr.2022.02.075. eCollection 2022 May.
Lymphangioleiomyomatosis is a rare multisystem disease associated with genetic mutations. The disease usually occurs in women of childbearing age and is characterized by infiltration of immature smooth muscle cells into the lungs, airways, and axial lymphatic systems of the chest and abdomen. The disease often destroys lung parenchyma and produces air cysts. Lymphangioleiomyomatosis cell infiltration of the lymphatic axis can affect hilar lymph nodes, mediastinal ganglia, and extrathoracic lymph nodes. The disease can cause lymphatic dilation in the lungs and thoracic ducts, causing chylous effusion into the pleural or abdominal cavities. Invasion of cells into the walls of pulmonary veins can lead to venous obstruction and pulmonary venous hypertension with hemoptysis. Most patients present with cough, dyspnea, pneumothorax, hemoptysis, and abnormal lung function. Definitive diagnosis is usually based on histopathology and immunohistochemistry. We present a case of LAM in a 36-year-old female patient who was confirmed by specimens obtained from pneumothorax surgery and positive immunohistochemical staining with HMB-45.
淋巴管平滑肌瘤病是一种与基因突变相关的罕见多系统疾病。该疾病通常发生在育龄女性,其特征是未成熟的平滑肌细胞浸润到肺、气道以及胸部和腹部的轴位淋巴系统。该病常破坏肺实质并产生气囊肿。淋巴管平滑肌瘤病细胞浸润淋巴轴可累及肺门淋巴结、纵隔神经节和胸外淋巴结。该病可导致肺部和胸导管的淋巴管扩张,引起乳糜液渗入胸腔或腹腔。细胞侵入肺静脉壁可导致静脉阻塞和肺静脉高压,并伴有咯血。大多数患者表现为咳嗽、呼吸困难、气胸、咯血和肺功能异常。确诊通常基于组织病理学和免疫组织化学。我们报告一例36岁女性淋巴管平滑肌瘤病患者,该病例通过气胸手术获取的标本及HMB - 45免疫组化染色阳性得以确诊。