Granulomatous cheilitis after treatment of venous malformations of the upper lip: a case report and literature review

Objective To explore the clinical manifestations, diagnosis, differential diagnosis, and treatment of granulomatous cheilitis complications after treatment in patients with upper lip venous malformations, as well as to provide a reference for their clinical diagnosis and treatment. Methods This report provides details on the clinical manifestations, diagnosis, differential diagnosis, and treatment of a case of granulomatous cheilitis after the treatment of upper lip venous malformation, and then analyzes granulomatous cheilitis alongside the related literature. The patient, a 30-year old male, was first seen in the dermatology department of another hospital with bright red spots on his lips, diagnosed with allergic dermatitis and received symptomatic treatment, and the erythema did not improve. He was diagnosed with ‘cavernous hemangioma’ and was treated with polydocanol and bleomycin injections. The bright red spots on his lips improved, but the swelling worsened for more than half a year. He then sought treatment at the oral mucosal department of our hospital. At the time of consultation, the swelling of the upper lip and perilabial skin was obvious, and there was a red patch on the right side of the upper lip, that was congested with blood. The upper lip was tough, with hard nodules, unclear borders, and poor mobility. Pathological examination showed epithelial hyperplasia of the upper lip mucosa, surface hyperkeratosis, subepithelial fibrous tissue hyperplasia, and chronic inflammation of the mucosa and minor salivary glands. Focal histiocyte, lymphocyte, and plasma cell infiltration was seen in the submucosal layer, with granulomatous inflammatory manifestations. Based on the patient's medical history, clinical manifestations, and histopathological manifestations, the diagnosis of granulomatous cheilitis was made. Tretinoin 0.3 mL (40 mg/mL, 1 mL/vial) was injected into the deep layer of the mucosa of the right and left upper lips for local block treatment. Prednisone acetate tablets (10 mg/Qd) were taken orally, and after 1 week of follow-up, the symptoms improved, so the original treatment was continued. After 2 weeks of follow-up, the swelling of the lips improved significantly, and the oral prednisone acetate tablets were adjusted to 5 mg/Qd. After 4 weeks of follow-up, the shape of the lips was largely back to normal, and the color and suppleness of the lips had improved significantly. The local block treatment and oral medication were stopped, and the patient was instructed to apply the topical tretinoin ointment Bid on the upper lip. Results The patient had a follow-up visit 8 weeks later, at which their lip color, shape, and texture remained normal, and the patient was instructed to stop the medication and follow up. A review of the literature suggests that the etiology of granulomatous cheilitis is unknown and that it is associated with genetic predisposition, odontogenic infections, allergic factors, microbial infections, and immunological factors. It needs to be clinically differentiated from diseases such as lip venous malformations, lip angioneurotic oedema, Crohn's disease, and tuberculosis. At present, the clinical treatment of granulomatous cheilitis is still based on local glucocorticoid block therapy or a combination of oral glucocorticoid drugs. In this case, the area of erythema on the lips decreased in size, but swelling occurred and continued to worsen after polydocanol and bleomycin injection treatment. Pre-existing venous malformation should be considered as a complication associated with injectable drugs that can produce granulomatous cheilitis. Conclusion The injection-based treatment of lip venous malformation may be complicated by granulomatous cheilitis, and in the process of clinical diagnosis and treatment, it is necessary to be aware to the existence of drug-related factors in the occurrence and development of granulomatous diseases.