Recurrent febrile lymphadenopathy treated with large doses of vitamin B1: report of two cases.

The 2 children whose cases are reported here both had recurrent episodes of fever and cervical lymphadenopathy. The conventional approach had been unsuccessful in identifying the cause or therapy. In neither case was there an infectious agent demonstrated, and biopsy of a pathologically enlarged lymph gland revealed only reactive hyperplasia in each case. Abnormal metabolism was revealed in the first patient by detecting a substance in urine which is reported to be diagnostic for a form of subacute necrotizing encephalomyelopathy. In the second case, red cell transketolase indicated thiamine pyrophosphate deficiency. Both children had elevated concentrations of folate and B12 in serum. Neither of the 2 patients had further episodes when given a clinical trial with large doses of thiamine hydrochloride. Recurrent episodes of febrile lymphadenopathy are extemely frequent in children and spontaneous resolution occurs, while in others there is either proven or assumed infection. Although final proof of therapeutic efficacy is lacking, the rapid improvement and maintenance of health in both children was striking after conventional therapy had failed.