Key result
Oral mexiletine dramatically relieves burning pain and soaking episodes in two children with primary erythromelalgia.
Why the study?
Primary erythromelalgia in children causes severe burning pain refractory to multiple pain medications, and effective treatment options are limited.
Does oral mexiletine improve symptoms in children with primary erythromelalgia?
Case Report (n=2)
Does oral mexiletine improve symptoms in children with primary erythromelalgia?
Oral mexiletine may be an effective treatment for refractory primary erythromelalgia in children.
May support mexiletine trial in refractory pediatric erythromelalgia; hypothesis-generating and requires controlled validation before practice change.
Primary erythromelalgia is characterized by burning pain, redness, and warmth in the extremities. We present two cases of primary erythromelalgia both of whom presented with a history of several months of severe burning pain in both hands and feet. Both patients had received multiple pain medications with no improvement in symptoms. Pain was relieved by putting affected parts in ice cold water, which resulted in immersion injury of the affected parts. Both patients stopped taking part in school and social activities. We tried oral mexiletine, a class Ib antiarrythmic agent, in view of its reported role in various chronic painful conditions. Dramatic improvement was observed with its use. Both patients improved after several weeks of use, and there were fewer soaking episodes. We observed no adverse effects with mexilitine therapy.
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Iqbal et al. (2009) conducted a case report in Primary erythromelalgia (n=2). Oral mexiletine was evaluated on Improvement in symptoms (pain relief, fewer soaking episodes). Oral mexiletine resulted in dramatic improvement of severe burning pain and fewer soaking episodes in two children with primary erythromelalgia, with no observed adverse effects.
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