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Journal of Islamic Dental Association of IRAN (JIDAI) / Summer 2012 /24 / (3) Javadzadeh et. al

nerve palsy [3]. Also in 1931, the term–Melkers- in its clinical features, with lips being the most
son- Rosenthal Syndrome was defined by Ro- common site of involvement. The frequent ma-
senthal to describe the triad of persistent lip and nifestation of the lesion is indicated as recurrent
face swelling, facial nerve palsy and fissured labial swellings that have the ability to remain
tongue [4]. In 1945, granulomatous lesion of the persistent [1-9]. The swelling is non-tender in
lip which was characterized by persistent lip palpation and is initially soft and non-pitting and
swelling (one sign of the Melkersson- Rosenthal later becomes rubbery and firm. Other oral ma-
Syndrome) was designated by Meischer as nifestations include: oral ulcers, submucoal
Meischer’s cheilitis [5]. Oral granulomatoses swellings, mucosal tags, fissured tongue (lingua
were described in accordance with some system- plica), angular cheilitis, gingival overgrowth,
ic conditions such as tuberculosis in 1951 [6], facial swelling and/or erythema, facial nerve pal-
sarcoidosis in 1985 [1] and Crohn’s disease in sy, and cervical lymphadenopathy.
2000 [7] But, the term orofacial granulomatosis Lip overgrowth (labial swelling)
or oro-facial granulomatosis was first presented Labial overgrowth can involve lower or upper
as a scientific term by eldWiesenf in 1985 that lip or both [10]. The swelling is often persistent
encompasses Melkersson-Rosenthal Syndrome but can also be recurrent, persisting for several
cheilitis granulomatosis of Meischer.(Fig.1) weeks or months [11]. The swelling may cause
Definition: Orofacial granulomatosis is used to enlargement or clefting of the lip(s) (median
describe non-caseous granulomatous inflamma- cheilitis) and inflammation and clefting of the
tion of the oral and facial region with recurrent corners of the mouth. (angular cheilitis) The
and persistent labial swellings in the absence of labial swelling is non-pitting at pressure and
any systemic disease. This lesion can be accom- non-tender in palpation and can vary from a soft
panied by manifestations such as oral ulcers, to a rubbery consistency based on its persistence.
gingival overgrowths, and cobblestone appear- The labial mucosa can be erythematous and have
ance of buccal mucosa [2]. In addition, forma- a granular appearance [9-12].
tion of granuloma results in obliteration of lym- Oral ulcers: The three principal types of ulcers
phatic vessels, formation of lymphedema, accu- can be encountered in orofacial granulomatosis
mulation of interstitial fluid and finally swelling with their most common feature of chronicity.
of the lips and other parts of the face. Orofacial In majority of cases, the ulcers are linear and
granuloma encompasses conditions previously longitudinal at the depth of the labial or buccal
referred to as Melkersson-Rosenthal Syndrome vestibule with exophytic margins with often ery-
and Meischer’s cheilitis. Orofacial granuloma is thematous borders [13-15].
an uncommon phenomenon, but diagnosis of The second less common type of ulcers are su-
new cases is currently increasing [1,2]. Contro- perficial symmetrical aphthous-like ulcers with
versies have recently been arisen about the point well circumscribed borders that can appear in
that whether orofacial granuloma is a distinct any part of oral mucosa.
clinical entity of a clinical manifestation of cer- The other type of ulcers which are associated
tain granulomatous diseases such as Crohn’s with orofacial granulomatosis are ulcers in the
disease or sarcoidosis [8]. In addition, other dis- real sense of the word but are described as pus-
orders such as profound fungal infections, tuber- tules in anterior part of the gingiva, labial vesti-
culosis, allergic angioedema, leprosy, Wegener’s bular mucosa or soft palate. They have the same
granuloma, etc. also exist with the same clinical appearance as pyostomatitis vegetans and are not
features and are later discussed in differential clinically purulent. In fact the term pustule is
diagnosis. used to describe them due to the appearance of
Clinical features: Orofacial granuloma I variable intraepithelial leukocytes in their microscopic

112 Summer 2012; Vol. 24, No. 3