Luo Di-Qing, Zhao Yu-Kun, Xu Qing-Fang, He Xiang-Qun, Wu Liang-Cai
Department of Dermatology, Huangpu Hospital of The First Affiliated Hospital.
Pain Med. 2014 Jun;15(6):1007-10. doi: 10.1111/pme.12343. Epub 2014 Jan 16.
Erythromelalgia is a rare clinical syndrome characterized by episodic attacks of burning pain, erythema, and increased temperature, primarily affecting the extremities, and in rare instances, involving the ear, face, neck, and the scrotum. The dermatoscopic features of erythromelalgia in a case with solely facial involvement have never been described previously.
We describe a 14-year-old female who presented with erythema, burning sensation, and warmth on her face only, which mimic the features of erythromelalgia. Physical examination showed higher temperature on the involved cheeks than on axillas during the episode, while the temperature on both areas was the same between episodes. Dermatoscope showed more dilated vessels inside the erythema during the episodes than between the episodes. The symptoms had excellent response to the combination treatment of gabapentin, indomethacin, and topical lidocaine compounds.
The present case is considered to be a variant of erythromelalgia. Its erythema may be resulted from the dilated vessels. Combination of modalities may provide effective management for erythromelalgia. "Erythermalgia" may be better than "erythromelalgia" to describe such conditions.
红斑性肢痛症是一种罕见的临床综合征,其特征为发作性灼痛、红斑和体温升高,主要累及四肢,极少数情况下累及耳部、面部、颈部和阴囊。此前从未描述过仅累及面部的红斑性肢痛症的皮肤镜特征。
我们描述了一名14岁女性,仅面部出现红斑、烧灼感和发热,类似于红斑性肢痛症的特征。体格检查显示,发作期间受累脸颊的温度高于腋窝,而发作间期两个部位的温度相同。皮肤镜检查显示,发作期间红斑内扩张的血管比发作间期更多。症状对加巴喷丁、吲哚美辛和外用利多卡因化合物的联合治疗反应良好。
本病例被认为是红斑性肢痛症的一种变体。其红斑可能由血管扩张引起。联合治疗方式可能为红斑性肢痛症提供有效的治疗。用“红斑热痛症”来描述这种情况可能比“红斑性肢痛症”更好。