Successful Treatment of Diffuse Pulmonary Lymphangiomatosis With Bevacizumab
Authors: Jurjan Aman, Erik Thunnissen, Marinus A. Paul, Geerten P. van Nieuw Amerongen, Anton Vonk-Noordegraaf
Journal: Annals of Internal Medicine (2012)

Letters5 June 2012Successful Treatment of Diffuse Pulmonary Lymphangiomatosis With BevacizumabJurjan Aman, MD, Erik Thunnissen, MD, PhD, Marinus A. Paul, MD, PhD, Geerten P. van Nieuw Amerongen, PhD, and Anton Vonk-Noordegraaf, MD, PhDJurjan Aman, MDFrom VU University Medical Center, Amsterdam, the Netherlands., Erik Thunnissen, MD, PhDFrom VU University Medical Center, Amsterdam, the Netherlands., Marinus A. Paul, MD, PhDFrom VU University Medical Center, Amsterdam, the Netherlands., Geerten P. van Nieuw Amerongen, PhDFrom VU University Medical Center, Amsterdam, the Netherlands., and Anton Vonk-Noordegraaf, MD, PhDFrom VU University Medical Center, Amsterdam, the Netherlands.Author, Article, and Disclosure Informationhttps://doi.org/10.7326/0003-4819-156-11-201206050-00016 SectionsAboutFull TextPDF ToolsAdd to favoritesDownload CitationsTrack CitationsPermissions ShareFacebookTwitterLinkedInRedditEmail Background: Diffuse pulmonary lymphangiomatosis is a rare disease caused by uncontrolled proliferation of lymphatic vessels. Although histologically benign, it can lead to death. It is distinct from lymphangiectasis and lymphangioleiomyomatosis but is often misdiagnosed. Treatment options are limited and often ineffective (1, 2).Objective: To report successful treatment of lymphangiomatosis with bevacizumab, an antibody that blocks vascular endothelial growth factor (VEGF).Case Report: In 2005, a 40-year-old woman presented to a hospital with chest pain and fatigue, and the physicians referred her to our hospital when they found a mediastinal mass and lesions in her spleen. Surgical exploration of her ...References1. Faul JL, Berry GJ, Colby TV, Ruoss SJ, Walter MB, Rosen GD, et al. Thoracic lymphangiomas, lymphangiectasis, lymphangiomatosis, and lymphatic dysplasia syndrome. Am J Respir Crit Care Med. 2000;161:1037-46. [PMID: 10712360] CrossrefMedlineGoogle Scholar2. Tazelaar HD, Kerr D, Yousem SA, Saldana MJ, Langston C, Colby TV. Diffuse pulmonary lymphangiomatosis. Hum Pathol. 1993;24:1313-22. [PMID: 8276379] CrossrefMedlineGoogle Scholar3. Tammela T, Alitalo K. Lymphangiogenesis: molecular mechanisms and future promise. Cell. 2010;140:460-76. [PMID: 20178740] CrossrefMedlineGoogle Scholar4. Wuest TR, Carr DJ. VEGF-A expression by HSV-1-infected cells drives corneal lymphangiogenesis. J Exp Med. 2010;207:101-15. [PMID: 20026662] CrossrefMedlineGoogle Scholar5. Ozeki M, Fukao T, Kondo N. Propranolol for intractable diffuse lymphangiomatosis [Letter]. N Engl J Med. 2011;364:1380-2. [PMID: 21470038] CrossrefMedlineGoogle Scholar Author, Article, and Disclosure InformationAuthors: Jurjan Aman, MD; Erik Thunnissen, MD, PhD; Marinus A. Paul, MD, PhD; Geerten P. van Nieuw Amerongen, PhD; Anton Vonk-Noordegraaf, MD, PhDAffiliations: From VU University Medical Center, Amsterdam, the Netherlands.Acknowledgment: The authors thank Dr. Jackeline Agorreta, University of Navarra, Pamplona, Spain, for her contribution.Disclosures: None disclosed. 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