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Successful Treatment of Diffuse Pulmonary Lymphangiomatosis With Bevacizumab

Jurjan Aman, MD; Erik Thunnissen, MD, PhD; Marinus A. Paul, MD, PhD; Geerten P. van Nieuw Amerongen, PhD; and Anton Vonk-Noordegraaf, MD, PhD
[+] Article and Author Information

From VU University Medical Center, Amsterdam, the Netherlands.

Acknowledgment: The authors thank Dr. Jackeline Agorreta, University of Navarra, Pamplona, Spain, for her contribution.

Potential Conflicts of Interest: None disclosed.


Ann Intern Med. 2012;156(11):839-840. doi:10.7326/0003-4819-156-11-201206050-00016
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Figure.

Treatment and characterization of diffuse pulmonary lymphangiomatosis.

A. Treatment effects of bevacizumab on blood hemoglobin levels. Bevacizumab was given every 3 wk, indicated by asterisks. B. The extent of lymphangiomatosis in the left lung as visualized on chest CT 1 day before (left) and 10 weeks after (right) initiation of bevacizumab treatment. C. Immunohistochemical staining of VEGF-A in proliferative lymphatic vessels (top) vs. unaffected lymphatic vessels from the same area (bottom) (original magnification, ×5). The brown VEGF-A staining is evident in lymphendothelial cells of proliferative lymphangioma vessels (top, inset) (original magnification, ×40) but almost absent in lymphendothelial cells of the vas afferens in a lymph node (bottom, inset) (original magnification, ×40). In addition, the density of lymphendothelial cells is higher in lymphangioma vessels than in the vas afferens, suggesting proliferation of lymphendothelial cells. The lymphatic character of the proliferative vessels was confirmed by staining for VEGFR-3 (data not shown). CT = computed tomography; VEGF = vascular endothelial growth factor.

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