M. Hino et al., Bone resorption associated with uncoupling of osteoclastic and osteoblastic activities in adult T cell leukemia with hypercalcemia: case report, ANN HEMATOL, 80(7), 2001, pp. 426-429
A 64-year-old woman with adult T cell leukemia (ATL) was admitted to our ho
spital with severe hypercalcemia. The serum calcium level was elevated to 1
4.9 mg/dl. Biochemical parameters for bone formation including serum osteoc
alcin (bone G1a protein, BGP) and alkaline phosphatase (ALP) were normal. T
he serum levels of tartrate-resistant acid phosphatase (TRAP), a parameter
for bone resorption, were increased (4.6 KAU). The serum level of parathyro
id hormone-related protein (PTHrP) was elevated (343 pmol/l). The cytokines
with stimulatory effects on bone resorption, such as interleukin (IL)-1 al
pha, IL-1 beta, IL-6, and tumor necrosis factor-alpha, were not detected. S
erum Ca levels, PTHrP levels, and TRAP levels decreased with the decrease i
n ATL cells after chemotherapy, while serum BGP levels and ALP levels incre
ased. On the 29th hospital day, ATL cells began to increase again. Then ser
-um PTHrP levels, Ca levels, and TRAP levels increased, while serum BGP lev
els and ALP levels decreased. A marked excessive bone resorption with suppr
essed bone formation (uncoupling) occurred in this patient. The ATL cells p
roduced not only PTHrP but also IL-1 alpha and IL-1 beta. These results sug
gest that PTHrP may act as a humoral factor and IL-1 may act as a local fac
tor in bone metabolism of ATL patients.