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[淋巴管平滑肌瘤病:病理生理学、诊断及治疗的最新进展]

[Lymphangioleiomyomatosis: Update on Pathophysiology, Diagnosis, and Treatment].

作者信息

Lagos Castillo Macarena, Reyes Cartes Felipe

机构信息

Instituto Nacional del Tórax, Santiago, Chile.

出版信息

Rev Med Chil. 2025 Jan;153(1):63-71. doi: 10.4067/s0034-98872025000100063. Epub 2025 Mar 20.

DOI:10.4067/s0034-98872025000100063
PMID:40214022
Abstract

Interstitial lung diseases (ILDs) include a group of rare and complex pathologies, among which lymphangioleiomyomatosis (LAM) stands out. LAM is considered a neoplastic disease characterized by abnormal proliferation of smooth muscle cells in the lung, forming cysts and causing severe respiratory alterations. LAM primarily affects women of childbearing age and can present in two forms: sporadic (S-LAM) and associated with tuberous sclerosis complex (TSC-LAM). Although its etiopathogenesis is not fully understood, genetic and hormonal pathophysiological mechanisms have been studied. Genetic alterations in the TSC1 and TSC2 genes lead to uncontrolled activation of the mTOR pathway, resulting in the transformation of smooth muscle cells into abnormal cells known as LAM cells. The activation of hormonal receptors present in LAM cells increases cell proliferation and migration. The perialveolar proliferation of these cells may explain alveolar rupture and cyst formation. There is also matrix remodeling and production of lymphangiogenic growth factors, such as vascular endothelial growth factor-D (VEGF-D), which contributes to the formation of lymphatic vessels in LAM lesions and cystic remodeling of the lung. The most common symptoms are exertional dyspnea and recurrent pneumothorax, along with other extrapulmonary manifestations such as renal angiomyolipomas. Diagnosis is based on characteristic tomographic findings and elevated plasma levels of VEGF-D. Treatment focuses on improving quality of life and halting disease progression. Sirolimus, an mTOR inhibitor, is the first-line therapy and has shown to stabilize lung function and reduce symptoms. In advanced cases, lung transplantation is a viable option. Hormone therapy and some surgical procedures are currently not recommended due to inconsistent results.

摘要

间质性肺疾病(ILDs)包括一组罕见且复杂的病理状况,其中淋巴管平滑肌瘤病(LAM)尤为突出。LAM被认为是一种肿瘤性疾病,其特征是肺内平滑肌细胞异常增殖,形成囊肿并导致严重的呼吸改变。LAM主要影响育龄女性,可表现为两种形式:散发性(S-LAM)和与结节性硬化症复合体相关(TSC-LAM)。尽管其发病机制尚未完全明确,但已对遗传和激素病理生理机制进行了研究。TSC1和TSC2基因的遗传改变导致mTOR通路的失控激活,从而使平滑肌细胞转化为称为LAM细胞的异常细胞。LAM细胞中存在的激素受体激活会增加细胞增殖和迁移。这些细胞的肺泡周围增殖可能解释了肺泡破裂和囊肿形成。还存在基质重塑和淋巴管生成生长因子的产生,如血管内皮生长因子-D(VEGF-D),这有助于LAM病变中淋巴管的形成和肺的囊性重塑。最常见的症状是劳力性呼吸困难和复发性气胸,以及其他肺外表现,如肾血管平滑肌脂肪瘤。诊断基于特征性的断层扫描结果和血浆VEGF-D水平升高。治疗的重点是改善生活质量并阻止疾病进展。西罗莫司,一种mTOR抑制剂,是一线治疗药物,已显示出可稳定肺功能并减轻症状。在晚期病例中,肺移植是一种可行的选择。由于结果不一致,目前不推荐激素治疗和一些外科手术。

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