Moreno Rodolfo P, Hernández Yanina, Garrido Patricia, Camargo Vargas Bethy, Hernández Alberto, Faín Javier, Seligra Cecilia, Topp Patricia, Maffey Alberto, Cardigni Gustavo
Servicio de Pediatría, Sanatorio de la Trinidad Palermo.
Cirugía Infantil, Sanatorio de la Trinidad Palermo.
Arch Argent Pediatr. 2021 Jun;119(3):e264-e268. doi: 10.5546/aap.2021.e264.
Diffuse pulmonary lymphangiomatosis is a rare disease characterized by marked proliferation and dilation of lymphatic vessels in the lungs, pleura, and mediastinum. The prevalence is unknown and the etiology is not fully understood. A 22-month-old girl was admitted for polyserositis, with pericardial and pleural effusion. She required pericardiocentesis and pleural drainage, presenting chyle drainage (1.5-4 liters/ day) without response to medical treatment (fasting, parenteral nutrition and octreotide). A lung biopsy was performed. The pathological anatomy showed findings compatible with diffuse pulmonary lymphangiomatosis. Treatment with sirolimus and propanolol began, decreasing losses due to pleural drainage one week after treatment. She progressed well, discontinued oxygen supply and pleural drainage was removed, leaving the patient after the fourth month of hospitalization. Early diagnosis of diffuse pulmonary lymphangiomatosis is difficult to achieve, but it allows the application of therapies that prevent disease progression, reducing morbidity and mortality.
弥漫性肺淋巴管瘤病是一种罕见疾病,其特征为肺、胸膜和纵隔内淋巴管显著增生和扩张。其患病率未知,病因也尚未完全明确。一名22个月大的女童因多发性浆膜炎入院,伴有心包和胸腔积液。她需要进行心包穿刺术和胸腔引流,出现乳糜引流(每天1.5 - 4升),药物治疗(禁食、肠外营养和奥曲肽)无效。进行了肺活检。病理解剖显示结果符合弥漫性肺淋巴管瘤病。开始使用西罗莫司和普萘洛尔治疗,治疗一周后胸腔引流导致的液体丢失减少。她病情进展良好,停止吸氧,拔除胸腔引流管,住院四个月后出院。弥漫性肺淋巴管瘤病的早期诊断难以实现,但它允许应用预防疾病进展的疗法,从而降低发病率和死亡率。