Nagy Stephanie, Fraser Marika, Kesselman Marc M
Department of Rheumatology, Kiran C. Patel College of Osteopathic Medicine, Nova Southeastern University, Davie, USA.
Division of Otolaryngology, Memorial Healthcare System, Hollywood, USA.
Cureus. 2025 Mar 20;17(3):e80879. doi: 10.7759/cureus.80879. eCollection 2025 Mar.
Cheilitis granulomatosa (CG) is a persistent and progressive swelling of the lips that can be non-tender and soft or firm to touch, with noncaseating granulomas that are perilymphatic and may show intralymphatic histiocytosis and lymphatic dilatation. CG can occur as an isolated condition or as part of Melkersson-Rosenthal syndrome, which also includes facial paralysis and a fissured tongue. The etiology of CG is currently unknown but has been hypothesized to be connected to genetics, allergies, immunological processes, and infectious causes. This case provides further evidence for the pathogenic causes of CG. We present a 46-year-old male patient with four years of progressive lip swelling to an outpatient rheumatological clinic, the cause of which has yet to be determined following numerous visits to healthcare providers. Corticosteroids were previously attempted, resulting in minor improvements in swelling; however, following discontinuation, the lip swelling returned. Laboratory findings were significant for Saccharomyces and Lyme disease, while other autoimmune biomarkers remained negative, and a biopsy indicated noncaseating granulomas, leading to the diagnosis of CG. The patient was started on mycophenolate, and following treatment, there was a significant reduction in the swelling of the lips. As the strongest cause of CG is currently unknown, this unique case brings awareness to the infectious causes leading to CG. It calls for a greater need to investigate the reason behind certain pathogens specifically targeting the lips without causing any other systemic effects.
肉芽肿性唇炎(CG)是一种唇部持续且进行性肿胀的疾病,触诊时可能无压痛,质地柔软或坚硬,伴有非干酪样肉芽肿,这些肉芽肿位于淋巴管周围,可能显示淋巴管内组织细胞增多和淋巴管扩张。CG可作为一种孤立的病症出现,也可作为梅尔克森 - 罗森塔尔综合征的一部分,该综合征还包括面瘫和沟纹舌。CG的病因目前尚不清楚,但据推测与遗传、过敏、免疫过程和感染因素有关。本病例为CG的致病原因提供了进一步的证据。我们介绍一位46岁男性患者,其唇部进行性肿胀四年,前往门诊风湿科就诊,在多次就诊于医疗服务提供者后,病因仍未确定。此前曾尝试使用皮质类固醇,肿胀有轻微改善;然而,停药后唇部肿胀又复发。实验室检查结果显示存在酿酒酵母和莱姆病,而其他自身免疫生物标志物仍为阴性,活检显示为非干酪样肉芽肿,从而诊断为CG。患者开始使用霉酚酸酯治疗,治疗后唇部肿胀明显减轻。由于目前CG最主要的病因尚不清楚,这个独特的病例让人们意识到导致CG的感染因素。它呼吁更有必要调查某些病原体专门靶向唇部而不引起任何其他全身影响的背后原因。