Intravascular papillary endothelial hyperplasia in the mandible: a case report

Abstract

Intravascular papillary endothelial hyperplasia (IPEH) is histopathologically characterized by papillary proliferation of vascular endothelial cells. IPEH in the mandible is very rare, such that only four affected patients have been described in the English-language medical literature. Thus, there is a poor understanding of the pathogenesis and clinical features of IPEH in the mandible. This case report describes a patient with IPEH in the mandible who had a history of repeated trauma involving the mandible due to boxing-related and baseball-related injuries. Imaging examinations had diagnostic limitations, in that they showed a multilocular radiolucency suggestive of a simple bone cyst of the mandible, whereas intraoperative findings revealed a fluid-free unicystic cavity lined by a thin red membrane. Thus, histopathologic examinations were necessary for definitive diagnosis. The specimen demonstrated a spongy structure consisting of many small papillary fibrous tissues, lined by a typical monolayer endothelium that expressed CD34, but did not express D2-40 or AE1/AE3. Moreover, the Ki-67 labeling index was <1%. Thus, the lesion was identified as intraosseous IPEH in the mandible. Although the pathogenesis of IPEH has been controversial, our findings in this case suggest that pathogenesis of IPEH may be related to a history of trauma.

Report of intraosseous intravascular papillary endothelial hyperplasia associated with an odontogenic cyst in the maxilla and literature review

“…In the present case, unlike other authors [ 12 – 16 ], we did not observe root resorption. However, areas of bone expansion, cortical resorption, maxillary sinus obliteration, and nasal septum destruction were noted.…”

“…Some immunohistochemical reactions may contribute to the diagnosis of IPEH, such as CD34 and vimentin [ 16 ]. Ki-67 may also help in cases where there is doubt in differentiating between IPEH and angiosarcoma, as the latter generally exhibits a high expression of Ki-67 [ 27 ].…”

Immunohistochemical Study of Differential Expressions of CD31, ERG, VEGF-A, α-SMA, Glut-1, CD105, D2-40 and Ki-67 in Oral Intravascular Papillary Endothelial Hyperplasia

“…In 1976, Clarkin and Enzinger 3) named this lesion "intravascular papillary endothelial hyperplasia", and this name has been used ever since. IPEH has the propensity to develop in the fingers, soles of the feet, and head and neck, while it is reported to be relatively rare in the oral cavity [4][5][6][7][8][9][10][12][13][14][15][16][17][18][19][20][21][22][23][24][25] . Lesions in the oral cavity are frequently reported in the lower lip, tongue, buccal mucosa, upper lip, and gingiva 5,26,27) .…”