A 14-year-old boy with longstanding multisystem Langerhans cell histiocytos
is (LCH) had multifocal bone pain unresponsive to chemotherapy, corticoster
oids, anti-inflammatories, and narcotic analgesics. He responded to two cyc
les of intravenous pamidronate, each 90 mg per day on three consecutive day
s. Two subsequent episodes of deterioration also responded to treatment. Th
e pathophysiology of his bone pain and the likely mechanisms of biphosphona
te action are discussed, with emphasis on the molecular and cellular basis
of LCH.