Abstract
We administered high doses of calcitriol (up to 32 μg per day) to an infant with malignant osteopetrosis, in an attempt to stimulate bone resorption. The patient was placed on a low-calcium diet to prevent hypercalcemia. Measures of bone turnover increased during calcitriol therapy; hydroxyproline excretion rose from 140 to 1358 μg per milligram of creatinine per 24 hours, with parallel increased in the ratio of calcium to creatinine in the urine, urinary gamma-carboxyglutamic acid, serum osteocalcin, and serum alkaline phosphatase. A pretreatment bone-biopsy specimen contained no osteoclasts with ruffled borders, a feature of active osteoclasts. After 11 days of calcitriol, ruffled borders were noted. After three months, numerous osteoclasts with ruffled borders and associated bony disruption were evident. Before therapy, the patient's monocytes were incapable of in vitro bone resorption, but after calcitriol, their resorptive capacity was increased to 3.3 times control levels. These data demonstrate that calcitriol increased bone mineral and matrix turnover in our patient. However, during the three months of calcitriol therapy there was only slight clinical improvement in her severe disease. Early and sustained treatment with calcitriol may be useful in osteopetrosis.
| Original language | English (US) |
|---|---|
| Pages (from-to) | 409-415 |
| Number of pages | 7 |
| Journal | New England Journal of Medicine |
| Volume | 310 |
| Issue number | 7 |
| State | Published - 1984 |
| Externally published | Yes |
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Treatment of congenital osteopetrosis with high-dose calcitriol. / Key, L.; Carnes, D.; Cole, S.; Holtrop, M.; Bar-Shavit, Z.; Shapiro, F.; Arceci, R.; Steinberg, J.; Gundberg, C.; Kahn, A.
In: New England Journal of Medicine, Vol. 310, No. 7, 1984, p. 409-415.Research output: Contribution to journal › Article
}
TY - JOUR
T1 - Treatment of congenital osteopetrosis with high-dose calcitriol
AU - Key, L.
AU - Carnes, D.
AU - Cole, S.
AU - Holtrop, M.
AU - Bar-Shavit, Z.
AU - Shapiro, F.
AU - Arceci, R.
AU - Steinberg, J.
AU - Gundberg, C.
AU - Kahn, A.
PY - 1984
Y1 - 1984
N2 - We administered high doses of calcitriol (up to 32 μg per day) to an infant with malignant osteopetrosis, in an attempt to stimulate bone resorption. The patient was placed on a low-calcium diet to prevent hypercalcemia. Measures of bone turnover increased during calcitriol therapy; hydroxyproline excretion rose from 140 to 1358 μg per milligram of creatinine per 24 hours, with parallel increased in the ratio of calcium to creatinine in the urine, urinary gamma-carboxyglutamic acid, serum osteocalcin, and serum alkaline phosphatase. A pretreatment bone-biopsy specimen contained no osteoclasts with ruffled borders, a feature of active osteoclasts. After 11 days of calcitriol, ruffled borders were noted. After three months, numerous osteoclasts with ruffled borders and associated bony disruption were evident. Before therapy, the patient's monocytes were incapable of in vitro bone resorption, but after calcitriol, their resorptive capacity was increased to 3.3 times control levels. These data demonstrate that calcitriol increased bone mineral and matrix turnover in our patient. However, during the three months of calcitriol therapy there was only slight clinical improvement in her severe disease. Early and sustained treatment with calcitriol may be useful in osteopetrosis.
AB - We administered high doses of calcitriol (up to 32 μg per day) to an infant with malignant osteopetrosis, in an attempt to stimulate bone resorption. The patient was placed on a low-calcium diet to prevent hypercalcemia. Measures of bone turnover increased during calcitriol therapy; hydroxyproline excretion rose from 140 to 1358 μg per milligram of creatinine per 24 hours, with parallel increased in the ratio of calcium to creatinine in the urine, urinary gamma-carboxyglutamic acid, serum osteocalcin, and serum alkaline phosphatase. A pretreatment bone-biopsy specimen contained no osteoclasts with ruffled borders, a feature of active osteoclasts. After 11 days of calcitriol, ruffled borders were noted. After three months, numerous osteoclasts with ruffled borders and associated bony disruption were evident. Before therapy, the patient's monocytes were incapable of in vitro bone resorption, but after calcitriol, their resorptive capacity was increased to 3.3 times control levels. These data demonstrate that calcitriol increased bone mineral and matrix turnover in our patient. However, during the three months of calcitriol therapy there was only slight clinical improvement in her severe disease. Early and sustained treatment with calcitriol may be useful in osteopetrosis.
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M3 - Article
C2 - 6546410
AN - SCOPUS:0021350813
VL - 310
SP - 409
EP - 415
JO - New England Journal of Medicine
JF - New England Journal of Medicine
SN - 0028-4793
IS - 7
ER -