Cukić Vesna, Lovre Vladimir, Beslić Serif, Bilalović Nurija, Guska Safet
Klinika za plućne bolesti i TBC Podhrastovi, Sarajevo.
Med Arh. 2008;62(3):180-2.
Lymphangioleiomyomatosis (LAM) is extreme rare diffuse lung disease of unknown cause seen almost exclusively in women of child-bearing age and rarely postmenopausal which indicates the involvement of hormones-estrogens. It results from proliferation of the cells having a smooth muscle cell phenotype (LAM cells) in the lung, and very often in the kidney and axial lymphatics and lymph nodes or any combination. It may occur sporadically or in association with the neurocutaneous syndrome--tuberous sclerosis. LAM cell proliferation may obstruct bronchioles, lymph vessels and venules that lead to airflow obstruction, formation of lung cysts, bullas and pneumothoraces, chylothorax, chylous ascites, hemosiderosis and hemoptysis. Approxymately 400 cases of LAM have been reported so far, most of them in USA. The average survival is about 8.5 years. There is no specific therapy. There are attempts with progesterone, lung transplantation, Doxycicline besides the symptomatic therapy. A new drug Rapamycin is tested. We are reporting 43-year old woman admitted in the Clinic for pulmonary diseases and TB "Podhrastovi" because of progressive dyspnea and suspect lung diffuse fibrosis, after the surgical treatment of spontaneous pneumothorax. The diagnosis of lung lymphangiomyomatosis was established by chest X ray, computerized chest tomography (CT), pathohistological findings of open lung biopsy. The treatment with progesterone is underway with other symptomatic therapy.
淋巴管平滑肌瘤病(LAM)是一种极为罕见的弥漫性肺部疾病,病因不明,几乎仅见于育龄期女性,绝经后女性罕见,提示激素——雌激素参与其中。它是由具有平滑肌细胞表型的细胞(LAM细胞)在肺部增殖引起的,常常还累及肾脏、纵隔淋巴管和淋巴结,或出现任何组合情况。它可散发,也可与神经皮肤综合征——结节性硬化症相关联。LAM细胞增殖可阻塞细支气管、淋巴管和小静脉,导致气流阻塞、肺囊肿、肺大疱和气胸形成,还可引起乳糜胸、乳糜腹水、含铁血黄素沉着症和咯血。迄今为止,大约已报告了400例LAM病例,其中大多数在美国。平均生存期约为8.5年。尚无特效疗法。除对症治疗外,有人尝试使用孕激素、肺移植、强力霉素。一种新药雷帕霉素正在进行试验。我们报告了一名43岁女性,因进行性呼吸困难和疑似肺弥漫性纤维化入住“Podhrastovi”肺病与结核病诊所,此前她接受了自发性气胸的手术治疗。通过胸部X线、胸部计算机断层扫描(CT)及开胸肺活检的病理组织学检查结果确诊为肺淋巴管平滑肌瘤病。目前正在进行孕激素治疗及其他对症治疗。