The online version of this article (doi:10.1186/1129-2377-15-18) contains supplementary material, which is available to authorized users.
Ming-Chun Chen, Qing-Fang Xu contributed equally to this work.
The authors declare that they have no competing interests.
CMC and XQF conceived of the study, participated in clinical management of the patient, reviewed the literature on the item and drafted the manuscript. LDQ made the correct diagnosis, participated in clinical management of the patient, conceived of the study, reviewed the literature on the item and drafted the manuscript. LX participated the clinical diagnosis, reviewed the literature, drafted and corrected the manuscript. HDY participated in the design of the paper, and drafted the manuscript. All authors read and approved the final manuscript.
Erythermalgia is a rare cutaneous disorder characterized by attacking of erythema, pain and increased temperature, which primarily involves the extremities and may infrequently extend to the neck, face, ears and even the scrotum. We reported an 18-year-old woman who presented with 3 years history of sole involvement of attacking erythema, pain and warmth over her face and ears without any other associations. The frequency and severity of the flares progressed gradually during the course. Cutaneous examination revealed erythema, increased temperature and tenderness on the face and ears during the flare. The symptoms could be relieved rapidly by cooling. Dermatoscope showed that vessels inside the erythema were more dilated during the episode than after application of ice. The lesion is considered a rare variant of erythermalgia with sole involvement of face and ears. The symptoms had mild response to oral antihistamines, topical steroids and tacrolimus, but had excellent response to the combinative therapy of aspirin and paroxetins.
Charles J, Buttaci DO: Erythromelalgia: a case report and literature review. Pain Med 2006, 7(6):534–538. 10.1111/j.1526-4637.2006.00240.x CrossRef
Johnson E, Iyer P, Eanes A, Zolnoun D: Erythema and burning pain in the vulva: a possible phenotype of erythromelalgia. Case Rep Med 2011. doi:10.1155/2011/374167
Luo DQ, Zhao YK, Xu QF, He XQ, Wu LC: Recurrent facial erythema with burning sensation and increased temperature: a variant of erythromelalgia or a new entity? Pain Med 2014. doi:10.1111/pme.12343
- Erythema associated with pain and warmth on face and ears: a variant of erythermalgia or red ear syndrome?
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