Annals of medicine and surgery (2012) · 2023
Pleural effusion due to lymphangiomatosis and the role of sirolimus: A case report.
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Riazuddin Muhammad, Farouk Noha I, Ali Saad S, Butt Muhammad I, Arabi Tarek Z, Sabbah Belal N, Ali Maha S, Alkattan Khaled
Аннотация
INTRODUCTION: Lymphangiomatosis is a rare abnormal proliferation of lymphatic vessels involving multiple organs like the brain, lung, heart, spleen, liver, and bones. Lymphangiomas constitute 5.6% of all benign tumors in infancy and adulthood.
CASE PRESENTATION: We report a case of a young lady who presented with constitutional symptoms and progressive dyspnea. Her medical history is significant for muco-cutaneous albinism, diffuse hemangiomas of the bone and viscera, and consumptive coagulopathy status post-splenectomy. After initial investigations, she was found to have right-sided pleural effusion. Pleural fluid analysis indicated chylothorax. She had multiple drainages of the pleural fluid done, and afterward, ligation of the right thoracic duct was performed with a trial of sirolimus, which improved her chylothorax.
CLINICAL DISCUSSION: Several case reports have reported positive outcomes with sirolimus in the treatment of lymphangiomatosis. However, larger controlled studies are needed to confirm these findings.
CONCLUSION: Sirolimus is promising as a medical treatment for diffuse pulmonary lymphangiomatosis.
CASE PRESENTATION: We report a case of a young lady who presented with constitutional symptoms and progressive dyspnea. Her medical history is significant for muco-cutaneous albinism, diffuse hemangiomas of the bone and viscera, and consumptive coagulopathy status post-splenectomy. After initial investigations, she was found to have right-sided pleural effusion. Pleural fluid analysis indicated chylothorax. She had multiple drainages of the pleural fluid done, and afterward, ligation of the right thoracic duct was performed with a trial of sirolimus, which improved her chylothorax.
CLINICAL DISCUSSION: Several case reports have reported positive outcomes with sirolimus in the treatment of lymphangiomatosis. However, larger controlled studies are needed to confirm these findings.
CONCLUSION: Sirolimus is promising as a medical treatment for diffuse pulmonary lymphangiomatosis.