Plasma cell mucositis: an unusual case

Abstract

A 36-year-old female Hepatitis B–positive patient was referred to the Oral Medicine Clinic at the Faculty of Dentistry, Chulalongkorn University, Bangkok in May 2017 complaining of severe oral cavity pain for over 5 months. She had a history of onset nonproductive cough since December 2016. She received no relief from Azithromycin, Codeine, or herbal lozenges. She reported that ulcerations developed on her right and left buccal mucosa 3 days after using the lozenges, thus, discontinued. Her physician prescribed prednisolone 10 mg 3x/day, which relieved her cough. However, her nocturnal coughing, burning sensation in her mouth, and dry lips persisted. During follow-up by her physicians in January 2017, she had conjunctivitis, generalized maculopapules on her extremities, dry scaly patches on her lips, multiple small red spots on the tip of the tongue, and ulcerative and erythematous patches on the left and right buccal mucosa that demonstrated pseudohyphae after a KOH 10% assay. Her chest radiograph revealed fibronodular infiltration in both upper lobes, complete blood count showed normal values except for a high eosinophil count (21%), and a 23 mm/h erythrocyte sedimentation rate (ESR) (normal 0–20 mm/h in women younger than 50 years old). Stool examination and urinary analysis appeared normal. Her anti-HIV and acid-fast bacteria (AFB) tests were negative. Clindamycin 300 mg was prescribed 3x/day for 7 days, and multivitamin, clotrimazole troche, triamcinolone acetonide 0.1%, and Lidocaine HCl gel. In February 2017, the burning sensation in her mouth worsened, with persistent oral lesions and palatal mucosa erosion. She developed discrete ill-defined blanchable erythematous rashes on her palms, soles, thighs, upper extremities, and upper chest. Additional investigations were performed including buccal mucosa biopsy. Histopathologically, the lesion demonstrated mucosal hyperplasia, no basal vacuolar change, and was nonmalignant. The laboratory results showed a 25% eosinophil count, 30 mm/h ESR, C-reactive protein (CRP) 3.85 (normal less than 10 mg/L), antinuclear antibody (ANA) positive with the titer of 1:320 (normal = negative) (speckled pattern), Anti-SSA/Ro‒positive with strong intensity, and Mycoplasma titer 1:80 (normal less than 1:40). The direct immunofluorescence, indirect immunofluorescence, Anti-dsDNA, Anti-La, Anti-Sm, Anti-nRNP, sputum and stool culture, AFB, and G6PD assays were negative. PUBLICATION HISTORY