Abstract

Although erythromelalgia (EM) has been documented in the literature for almost 150 years, it is still poorly understood. To overcome this limitation, we examined the spatial distribution of epidemic EM, and explored the association between temperature fluctuation and epidemic EM outbreaks in China. We searched all peer-reviewed literature on primary epidemic EM outbreaks in China. A two-stage model was used to characterize the relationship between temperature fluctuation and epidemic EM outbreaks. We observed that epidemic EM outbreaks were reported from 13 provinces during 1960–2014 and they mainly occurred between February and March in southern China. The majority of EM cases were middle school students, with a higher incidence rate in female and resident students. The major clinical characteristics of EM cases included burning, sharp, tingling and/or stinging pain in toes, soles and/or dorsum of feet, fever, erythema and swelling. A large “V”-shaped fluctuation of daily average temperature (TM) observed during the epidemic EM outbreaks was significantly associated with the number of daily EM cases (β = 1.22, 95%CI: 0.66 ~ 1.79), which indicated that this “V”-shaped fluctuation of TM probably triggered the epidemic EM outbreaks.

Highlights

  • Erythromelalgia (EM) has been documented in the literature for almost 150 years, it is still poorly understood

  • We examined the spatial distribution of epidemic EM, and explored the association between temperature fluctuation and epidemic EM outbreaks in China

  • Some studies found that an epidemic EM outbreak often followed a large ambient temperature fluctuation between February and March hypothesizing that the sharp drop and subsequent rapid rising of ambient temperature possibly induced the dysfunction of peripheral vasodilation and induced the EM28,29,38

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Summary

Introduction

Erythromelalgia (EM) has been documented in the literature for almost 150 years, it is still poorly understood. Between late February and early March of 2014, another epidemic EM outbreak with 494 cases was reported from two middle schools of Foshan city, Guangdong province, southern China. Some studies found that an epidemic EM outbreak often followed a large ambient temperature fluctuation between February and March hypothesizing that the sharp drop and subsequent rapid rising of ambient temperature possibly induced the dysfunction of peripheral vasodilation and induced the EM28,29,38 Until now this hypothesized relationship between temperature and epidemic EM has not been tested quantitatively, and some other questions about choice of temperature indicator (average temperature, maximum temperature, minimum temperature, or their corresponding apparent temperature) to best predict an EM outbreak have remained unanswered. Answering these questions will extend our understanding of the mechanisms of epidemic EM, and provide important information for the control and prevention of this disease in the future

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