A 5-year-old boy reacted from infancy to cold stimuli with severe local pain, widespread erythema, and occasionally muscle spasm and collapse. In early life intractable constipation was also a feature. Physical examination and routine laboratory studies were normal as were specialized studies on serum and urine. The ice test produced pain and erythema without urticaria and was blocked by previous infiltration with procaine hydrochloride. Intradermal injections of histamine and bradykinin produced typical reactions; serotonin produced marked erythema without a wheal. Injected plasma from cold-incubated, autologous blood also resulted in marked erythema. Passive transfer tests were negative. Therapeutic trial with several classes of compounds offered only slight relief. It is felt that this cold sensitivity may be mediated by local serotonin release.
Shelley WB, Caro WA. Cold Erythema: A New Hypersensitivity Syndrome. JAMA. 1962;180(8):639–642. doi:10.1001/jama.1962.03050210001001
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