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Polyglandular autoimmune syndrome type II associated with oral lichen planus
Author(s) -
Michele Davido Migngna,
Stefano Fedele,
Lucio Lo Russo,
Elvira Ruoppo,
Lorenzo Lo Muzio
Publication year - 2002
Publication title -
international journal of dermatology
Language(s) - English
Resource type - Journals
SCImago Journal Rank - 0.677
H-Index - 93
eISSN - 1365-4632
pISSN - 0011-9059
DOI - 10.1046/j.1365-4362.2002.14505.x
Subject(s) - medicine , tongue , glossitis , acanthosis , pathology , lingual papilla , biopsy , oral lichen planus , lesion , oral mucosa , dermatology , hyperkeratosis
A 42‐year‐old women was referred to our Division of Oral Medicine with a complaint of oral discomfort and of a red lesion of the dorsum of the tongue. Her past medical history revealed Addison’s disease, type I diabetes, hypothyroidism and noninfectious chronic hepatitis. On oral examination the patient showed a red atrophic rhomboid‐oval lesion in the dorsal midline of the tongue, just in front of the circumvallate papillae. The clinical appearance was that of median rhomboidal glossitis. A more complete intraoral examination revealed numerous interlacing keratotic striae in conjunction with atrophic and erosive areas localised bilaterally on the buccal mucosa ( Fig. 1). In addition, the upper vestibular mucosa showed erosive‐atrophic lesions mixed with small keratotic plaques. The clinical appearance was that of lichenoid lesion. Histologic examination of a buccal mucosa biopsy specimen revealed the presence of hyperorthokeratosis, acanthosis, vacualization of the basal cell layer, and an intense band‐like infiltration of lymphocytes ( Fig. 2). An incisional biopsy of the dorsal tongue was also taken, showing loss of filiform papillae, epithelial atrophy, and an underlying dense infiltrate of lymphocytes with a sharply demarcated lower margin. Routine laboratory investigations, including tests for hepatic and kidney functionality, markers of hepatitis A, B and C viruses, red and white blood cell count, platelet, ANA, were within normal limits. On the basis of these findings and with regard to the history of endocrine polyglandular hypofunction, a diagnosis of oral lichen planus (OLP) associated with polyglandular autoimmune syndrome type II was made. The patient was treated with high‐potency topical steroids in an adhesive base. An induction therapy of two applications daily for 1 month and a maintenance therapy of one application daily for 20 days was performed. Antimycotic mouthwashes were used in order to prevent oral candidiasis. The patient has recovered completely with the total disappearance of the ulcerative‐erosive lesions and relief of oral discomfort. In order to diagnose other component disorders of the syndrome and to prevent possible life‐threatening complications, a re‐evaluation of the patient will be taken repeatedly over time. Figure 1 Keratotic striae in conjuction with atrophic and erosive areas of the right buccal mucosa2Biopsy specimen from the right buccal mucosa shows hyperorto (para) keratosis, acanthosis, vacualization of the basal cell layer, and an intense band‐like infiltration of lymphocytes (hematoxylin and eosin stain, × 300)