TY - JOUR
T1 - 1α,25-dihydroxyvitamin D3 corrects osteomalacia in hypoparathyroidism and pseudohypoparathyroidism
AU - Epstein, S.
AU - Meunier, P. J.
AU - Lambert, P. W.
AU - Stern, P. H.
AU - Bell, N. H.
PY - 1983
Y1 - 1983
N2 - Deficiency of circulating 1α,25-dihydroxy-vitamin D (1α,25(OH)2D) regularly occurs in hypoparathyroidism (HP) and pseudohypoparathyroidism (PHP). Osteomalacia is occasionally found in the two diseases. Two patients, one with HP and the ther with PHP, both with symptomatic and biopsy-proven osteomalacia, were studied before and after treatment with 1α,25(OH)2D3. Laboratory values before treatment were as follows: serum immunoreactive parathyroid hormone was undetectable in the patient with HP and was elevated in the patient wth PHP. Serum 25-hydroxyvitamin D, measured by binding assay, was 131.5 and 61.9 nmol/l (normal: 69.1 ± 15.9 nmol/l); serum 24,25-dihydroxy-vitamin D, measured by binding assay, was 13.9 and 3.8 nmol/l (normal: 3.4 ± 1.4 nmol/l); serum 1α,25(OH)2D, measured by bioassay, was 28.6 and 29.0 pmol/l (normal: 77.3 ± 22.8 pmol/l) and, measured by receptor assay, was 36.2 and 41.0 pmol/l (normal: 71.8 ± 35.8 pmol/l) in the HP and PHP patients, respectively. Serum calcium was low and serum inorganic phosphate was high in both cases. Treatment with 1α,25(OH)2D3 (3-5 μg per day for 10-12 months) restored serum calcium and inorganic phosphate to normal, alleviated bone pain and healed the osteomalacia as shown on repeat bone biopsy. Our results provide further evidence that isolated deficiency of 1α,25(OH)2D may cause osteomalacia or rickets.
AB - Deficiency of circulating 1α,25-dihydroxy-vitamin D (1α,25(OH)2D) regularly occurs in hypoparathyroidism (HP) and pseudohypoparathyroidism (PHP). Osteomalacia is occasionally found in the two diseases. Two patients, one with HP and the ther with PHP, both with symptomatic and biopsy-proven osteomalacia, were studied before and after treatment with 1α,25(OH)2D3. Laboratory values before treatment were as follows: serum immunoreactive parathyroid hormone was undetectable in the patient with HP and was elevated in the patient wth PHP. Serum 25-hydroxyvitamin D, measured by binding assay, was 131.5 and 61.9 nmol/l (normal: 69.1 ± 15.9 nmol/l); serum 24,25-dihydroxy-vitamin D, measured by binding assay, was 13.9 and 3.8 nmol/l (normal: 3.4 ± 1.4 nmol/l); serum 1α,25(OH)2D, measured by bioassay, was 28.6 and 29.0 pmol/l (normal: 77.3 ± 22.8 pmol/l) and, measured by receptor assay, was 36.2 and 41.0 pmol/l (normal: 71.8 ± 35.8 pmol/l) in the HP and PHP patients, respectively. Serum calcium was low and serum inorganic phosphate was high in both cases. Treatment with 1α,25(OH)2D3 (3-5 μg per day for 10-12 months) restored serum calcium and inorganic phosphate to normal, alleviated bone pain and healed the osteomalacia as shown on repeat bone biopsy. Our results provide further evidence that isolated deficiency of 1α,25(OH)2D may cause osteomalacia or rickets.
UR - http://www.scopus.com/inward/record.url?scp=0020598454&partnerID=8YFLogxK
UR - http://www.scopus.com/inward/citedby.url?scp=0020598454&partnerID=8YFLogxK
U2 - 10.1530/acta.0.1030241
DO - 10.1530/acta.0.1030241
M3 - Article
C2 - 6687971
AN - SCOPUS:0020598454
VL - 103
SP - 241
EP - 247
JO - Free Radical Biology and Medicine
JF - Free Radical Biology and Medicine
SN - 0891-5849
IS - 2
ER -