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Research Letter |

Expression of Phosphodiesterase-5 in Lymphatic Malformation Tissue

Julie S. Green, MD, PhD1; Lori Prok, MD1; Anna L. Bruckner, MD1
[+] Author Affiliations
1Department of Dermatology, University of Colorado School of Medicine, Aurora
JAMA Dermatol. 2014;150(4):455-456. doi:10.1001/jamadermatol.2013.7002.
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Lymphatic malformations (LMs) are uncommon and sometimes debilitating congenital vascular anomalies that presumably arise because of developmental dysplasia of the lymphatic network in utero. They comprise primitive lymphatic sacs surrounded by a thickened layer of connective tissue and interspersed muscle fibers.1 Current treatments for LM are palliative and only partially successful and include compression, surgical resection, laser ablation, and sclerotherapy. A recent report by Swetman and colleagues2 noted marked reductions in LM size in 3 children after starting treatment with oral sildenafil citrate, an inhibitor of phosphodiesterase isoform 5 (PDE5). The localization of PDE5 in LM tissue and the mechanism of the effect of sildenafil on LM is unknown. To further investigate PDE5 localization within LM tissue, we performed immunohistochemical studies to identify the presence and relative location of PDE5 expression in vascular smooth muscle, vascular endothelium, and lymphatic endothelium.

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Figure.
Expression of CD34, D240, and Phosphodiesterase-5 (PDE5) in Lymphatic Malformation Tissue

A, Representative section of lymphatic malformation tissue showing dilated cystic spaces filled with proteinaceous fluid (hematoxylin-eosin, original magnification ×4); B, CD34 (red) localizes to perivascular endothelial cells (immunohistochemical stain, original magnification ×20); C, D240 (brown) is expressed in lymphatic endothelium (immunohistochemical stain, original magnification ×20); D, PDE5 (red) was seen in perivascular smooth muscle adjacent to lymphatic spaces (immunohistochemical stain, original magnification ×20).

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