Effect of Long-Term Calcitonin Therapy on the Clinical Course of Osteogenesis Imperfecta

Abstract

10 children with osteogenesis imperfecta, 4 with "tarda" and 6 with "congenita" varieties of the disease, were treated with salmon calcitonin (SCT) for intervals ranging from 14 to 35 months. Responses to SCT therapy in patients with osteogenesis imperfecta tarda were characterized by an apparent decreased fracture incidence in three, a fall in either alkaline or acid phosphatase, and a rate of increase in forearm bone mass which was greater than that observed in an untreated "tarda" population. The chemical response SCT therapy varied in children with osteogenesis imperfecta congenita, only one demonstrating a decrease in both acid phosphatase and urinary hydroxyproline. Three others responded with a rise in acid phosphatase, two of whom also demonstrated a fall in urinary hydroxyproline; in two other "congenita" patients urinary hydroxyproline was actually higher after SCT treatment and acid phosphatase relatively unchanged. Alkaline phosphatase was normal in all "congenita" patients before and following the SCT treatment interval. These varied biochemical responses were associated with temporary increments in bone mass early in the treatment course, although in bone mass early in the treatment course, although one "congenita" patient with the largest calciuric response to SCT and an increase in hydroxyproline excretion demonstrated progressive increments in skeletal mineral content during a 14-month treatment interval. In both "tarda" and "congenita" subjects, parathyroid hormone was unchanged by chronic SCT treatment; SCT-antibodies were detectable although biological responsivity to SCT persisted.

Lack of effects of human calcitonin in osteogenesis imperfecta

“…In the present study, where the BMC values were used to determine any change in bone mass during the treatment period, only two patients revealed a significant reduction in BMC. In disagreement with Rosenberg et al (1977), we found no changes in BMC during calcitonin therapy.…”

“…Like others (Castells et al 1972,19761, we found no changes in serum calcium or phosphorus concentrations during treatment. Rosenberg et al (1977) found that long-term therapy with salmon calcitonin resulted in serum values for calcium and phosphorus which in most cases were lower than the pre-treatment values. In our study the 24-h excretion of hydroxyproline was normal, but at the end of the treatment period a slight increase in the hydroxyproline excretion was noted.…”

“…In our study the 24-h excretion of hydroxyproline was normal, but at the end of the treatment period a slight increase in the hydroxyproline excretion was noted. Elevated, normal and subnormal hydroxyproline excretions in patients with 01 have previously been reported, but in none of these studies has it been proved that 01 is predominantly a disorder with increased bone turnover (Caniggia & Gennari 1972, Castells et al 1972, Goldfield et al 1972, Riley et al 1973, Smith et al 1975, Rosenberg et al 1977.…”

“…Increased bone turnover characterizes Paget's disease of bone, and human calcitonin has proved effective in the treatment of this disease (MacIntyre 1976). A favorable treatment response has also been reported in patients with 0 1 using salmon or porcine calcitonin (Caniggia & Gennari 1972, Castells et al 1972, 1974, 1976, Goldfield et al 1972 Armstrong et al 1975, Rosenberg et al 1977). Therapeutic trials with human calcitonin in 0 1 have not yet been published.…”

Serum Calcitonin and Bone Mineral Content in Patients with Osteogenesis Imperfecta

“…Osteogenesis imperfecta is an infrequent generalized disease in bone and connective tissue characterized by increased bone resorption, osteopenia and spontaneous fractures (Rosenberg et al 1977). Both short-term and long-term studies of calcitonin treatment have suggested an improvement in bone changes in patients with osteogenesis imperfecta tarda (Rosenberg et al 1977, Castells et al 1972.…”