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Intralymphatic Granulomas as a Pathogenic Factor in Cheilitis Granulomatosa/Melkersson–Rosenthal Syndrome: Report of a Case With Immunohistochemical and Molecular Studies

González-García, Carmen MD*; Aguayo-Leiva, Ingrid MD; Pian, H MD*; Fernández-Guarino, Montserrat PhD; Jaén-Olasolo, Pedro PhD

The American Journal of Dermatopathology: August 2011 - Volume 33 - Issue 6 - p 594-598
doi: 10.1097/DAD.0b013e3181f04912
Extraordinary Case Report

Orofacial granulomatosis, an uncommon immunologically mediated disorder, includes cheilitis granulomatosa and Melkersson-Rosenthal syndrome. It is clinically characterized by recurrent or persistent swelling of the orofacial tissues with a spectrum of other orofacial features and sometimes with neurological symptoms. The pathological findings are varied but are often characterized by the presence of noncaseating granuloma. We present a new case of orofacial granulomatosis with unusual histopathological findings, namely, intralymphatic granulomas. These may be the cause of the tissue edema. We demonstrated, by immunohistochemical studies, the lymphatic nature of the vessels affected by the granulomatous process.

From the Departments of *Pathology; †Dermatology, Ramón y Cajal Hospital, University of Alcalá, Madrid, Spain.

Funding sources: None.

Conflicts of interest: None declared.

Reprints: Ingrid Aguayo-Leiva, MD, Department of Dermatology, Hospital Ramón y Cajal Ctra, Colmenar, km 9,100. 28034-Madrid, Spain (e-mail:

© 2011 Lippincott Williams & Wilkins, Inc.