Ciacci Carolina, Bucci Cristina, Zingone Fabiana, Iovino Paola, Amato Massimo
Gastroenterology Unit, Department of Medicine and Surgery, University of Salerno, Baronissi Campus, via S, Allende, 84081 Baronissi, Salerno, Italy.
J Med Case Rep. 2014 Nov 30;8:397. doi: 10.1186/1752-1947-8-397.
Cheilitis granulomatosa causes persistent idiopathic lip swelling and ulceration and it can sometimes be recognized as a unique or early manifestation of Crohn's disease. Spontaneous remission is rare and with the lack of controlled trials, different therapeutic approaches have been used. Some cases have been treated with an exclusion diet in the attempt to rule out diet allergens, while the most popular treatments include antibiotics such as tetracycline and clofazimine tranilast, benzocaine topical or intralesional steroids, and cheiloplasty, with different outcomes.
We describe the case of a 23-year-old Caucasian man, primarily diagnosed with cheilitis granulomatosa for a severe lower lip swelling, and then with Crohn's disease of the terminal ileum and anus. Treatment of Crohn's disease with an anti-tumor necrosis factor alpha agent (infliximab) successfully induced remission of both the gastrointestinal disease and the oral lesion.
Our recommendation is that physicians should be able to recognize cheilitis granulomatosa as a possible marker of a more complex systemic disease and proceed first with an accurate physical examination, and further suggest investigations of the bowel. In cases of Crohn's disease, a therapy with biological agents can be successful.
肉芽肿性唇炎会导致持续性特发性唇部肿胀和溃疡,有时可被视为克罗恩病的一种独特或早期表现。自发缓解较为罕见,由于缺乏对照试验,人们采用了不同的治疗方法。一些病例采用排除饮食法以排除饮食过敏原,而最常用的治疗方法包括使用抗生素(如四环素和氯法齐明)、曲尼司特、苯佐卡因外用或病灶内注射类固醇以及唇成形术,但疗效各异。
我们描述了一名23岁的白种男性病例,最初因严重的下唇肿胀被诊断为肉芽肿性唇炎,随后被诊断为回肠末端和肛门克罗恩病。使用抗肿瘤坏死因子α药物(英夫利昔单抗)治疗克罗恩病成功诱导了胃肠道疾病和口腔病变的缓解。
我们建议医生应能够将肉芽肿性唇炎识别为更复杂的全身性疾病的可能标志,并首先进行准确的体格检查,进一步建议对肠道进行检查。对于克罗恩病病例,使用生物制剂进行治疗可能会取得成功。