Kechichian Elio, Haber Roger, Mourad Nadim, El Khoury Rana, Jabbour Samer, Tomb Roland
Department of Dermatology, Hotel Dieu de France University Hospital, Beirut, Lebanon.
Faculty of Medicine, Saint Joseph University, Beirut, Lebanon.
Int J Dermatol. 2017 May;56(5):486-495. doi: 10.1111/ijd.13584. Epub 2017 Feb 23.
Pyoderma gangrenosum (PG) is a sterile neutrophilic disorder that rarely affects children. Clinical, epidemiological, and therapeutic data on pediatric PG is poor as there are many newly reported associated diseases and drugs. This paper aims to review all recent available data on pediatric PG. A systematic review of the literature was conducted using Embase, Medline, and Cochrane databases. A total of 132 articles were included in the review. The most commonly reported underlying diseases in pediatric PG are inflammatory bowel diseases followed by hematologic disorders, vasculitis, immune deficiencies and Pyogenic Arthritis, Pyoderma gangrenosum and Acne (PAPA) syndrome. More than half of the cases occur with no underlying disease. The most frequently reported clinical presentation is multiple disseminated ulcers. Treatment should be tailored according to the underlying etiology. It includes systemic steroids, corticosteroid sparing agents such as dapsone and cyclosporine, and TNF-alpha inhibitors such as adalimumab and infliximab. Response to treatment is high with cure rates reaching 90%. A high index of suspicion and a thorough workup are mandatory in the management of pediatric PG.
坏疽性脓皮病(PG)是一种罕见累及儿童的无菌性中性粒细胞性疾病。由于有许多新报道的相关疾病和药物,关于儿童PG的临床、流行病学和治疗数据较少。本文旨在综述有关儿童PG的所有最新可用数据。使用Embase、Medline和Cochrane数据库对文献进行了系统综述。该综述共纳入132篇文章。儿童PG中最常报道的基础疾病是炎症性肠病,其次是血液系统疾病、血管炎、免疫缺陷以及化脓性关节炎、坏疽性脓皮病和痤疮(PAPA)综合征。超过一半的病例无基础疾病。最常报道的临床表现是多发性播散性溃疡。治疗应根据潜在病因进行调整。治疗方法包括全身用类固醇、皮质类固醇节约剂如氨苯砜和环孢素,以及肿瘤坏死因子-α抑制剂如阿达木单抗和英夫利昔单抗。治疗反应良好,治愈率达90%。在儿童PG的管理中,高度的怀疑指数和全面的检查是必不可少的。