Hypothermia, bradycardia and hypotension during glucocorticoid or cyclosporine A therapy in a boy with Kikuchi-Fujimoto disease.

Abstract

Kikuchi-Fujimoto disease (KFD) is an inflammatory disease of unknown etiology characterized by fever and cervical lymphadenopathy. Although KFD is a self-limiting disease, patients with severe or long-lasting course require glucocorticoid (GC) therapy. We report a presently 17-year-old boy with KFD who had 7 relapses since the onset at 4-year-old. He suffered from hypothermia, bradycardia, and hypotension during the treatment with prednisolone or methylprednisolone. All of his vital signs recovered after cessation of the drug in addition to fluid replacement and warming. Thus, GC was effective but could not be continued because of the adverse event. Although hypothermia developed during the treatment with 5 mg/kg/day of cyclosporine A (CsA) at his second relapse, he was successfully treated with lower-dose CsA (3 mg/kg/day). Thereafter, he had five relapses of KFD until the age of 12 and was treated by 1.3-2.5 mg/kg/day of CsA. Hypothermia accompanied by bradycardia and hypotension developed soon after concomitant administration of ibuprofen at his 5th and 6th relapses even during low-dose CsA therapy. Conclusively, GC, standard dose of CsA or concomitant use of NSAIDs may cause hypothermia, bradycardia and hypotension and needs special attention. Low-dose CsA could be a choice for such cases with KFD.