IntroductionThe term orofacial granulomatosis was introduced to encompass the broad spectrum of non-necrotizing granulomatous inflammation in the oral and facial region, including patients with the complete triad of MelkerssonRosenthal syndrome (MRS), chelitis granulomatosa, sarcoidosis, Crohn's disease and infectious disorders such as tuberculosis (1,2). There may be several underlying etiologic mechanisms, with similar clinical and pathologic presentations, often manifesting at different points in time (3).Granulomatous glossitis was first described as a peculiar manifestation of MRS by Schuermann in 1952 (4). The typical triad of MRS is rarely seen simultaneously, and involvement of the tongue as a sole manifestation of MRS is much rarer. In monosymptomatic cases, making a clear diagnosis is difficult, and therefore a complete differential diagnosis for the other recurrent or persistent disorders characterized by macroglossia has to be done (5).Herein we report a case of granulomatous glossitis with no associated systemic disorder that was unresponsive to clofazimine and metronidazole, yet showed minor improvement with tetracycline.
Case ReportA 50-year-old man presented with a complaint of enlargement of the tongue associated with small glossal tags. The papules had developed at the edge of the tongue and gradually increased in number during the previous five years. The patient complained of speech impairment, hypersalivation and a burning sensation on eating. He denied swelling of the lips, buccal and labial mucosa, and the face. His medical history was unremarkable except for hypertension that had been controlled with anti-hypertensive drug therapy for the previous 10 years.Dermatologic examination revealed slight enlargement of the tongue with a deep central furrow, multiple shallow radial fissures and multiple white mucosal tags at the edge of the tongue (Figs. 1 and 2).Laboratory investigations including a complete blood count, erythrocyte sedimentation rate, serum electrolytes, hepatic and renal function tests, serum iron level, anti-HIV antibodies, angiotensin converting enzyme level, thyroid function tests, IgG, IgA, IgM, IgE levels, ANA, anti-DNA, 24 hours urine calcium level, and stool examination for the presence of blood were all either negative or within normal limits.