An Unusual Case of Cheilitis Glandularis

Cheilitis glandularis (CG), a rare inflammatory disease, affects minor salivary glands, usually in the lower labial mucosa. Objective: We present a case of a 73 y/o male with multiple painless swellings located in the upper labial mucosa, in which two were purulent. The patient reported smoking four packs of cigarettes a day for 30 years, chronic exposure to sunlight, and a similar presentation in his sibling. Methods: The clinical approach included antibiotics followed by excisional biopsies of the purulent lesions. Results: Histopathologic findings were non-specific. However they were consistent with a diagnosis of suppurative CG as described in the literature. The patient remained under supervision and had no additional purulent lesions after six months. Conclusions: CG is a rare condition, with less than 40 cases reported in the English literature, and only 7 affected the upper lip. It is considered a hereditary autosomal dominant disease and middle aged men are more likely to be affected. The exact etiology is unknown but chronic sunlight exposure, smoking, poor oral hygiene, wind, and a compromised immune system are predisposing factors. The three types of CG (simple, superficial suppurative and deep suppurative) most likely represent progressive developmental stages of the same pathologic condition. Clinical management is primarily based on the clinical presentation and includes antibiotic therapy for infected lesions and avoidance of exposure to the predisposing factors. Surgical removal of the lesions is also recommended. Histopathologic findings are generally non-specific and include ductal ectasia, oncocytic metaplasia of ductal lining and sialadenitis. A possibility of malignant transformation to squamous cell carcinoma has been reported, essentially associated with the deep suppurative type of lesions. Therefore, close follow-up is mandatory.