vitamin-d-2 and Hypoparathyroidism

Hypoparathyroidism (HypoPT) is a rare endocrine disorder characterized by the absence or insufficient parathyroid hormone production resulting in chronic hypocalcemia. Complications of HypoPT include perturbation of several target organs. The conventional treatment consists of the administration of active vitamin D, namely calcitriol. Regarding vitamin D status, few data are available, mostly in HypoPT subjects supplemented with parent vitamin D. In addition, perturbation of vitamin D metabolism has been poorly investigated, as well as the contribution of altered vitamin D status on the clinical expression of the disease. The most recent consensus on the management of chronic HypoPT suggests the baseline evaluation of serum 25-hydroxy-vitamin D [25(OH)D] and supplementation with parent vitamin D with the aim to achieve and maintain serum 25(OH)D levels in the range of 30-50 ng/mL. The rationale for using supplementation with parent vitamin D (either ergocalciferol or cholecalciferol) in HypoPT would be to provide sufficient 25(OH)D substrate to the residual 1-α-hydroxylase activity, thus ensuring its conversion to active vitamin D in renal and extra-renal tissues. More data from experimental and clinical studies are needed for better assessing how these mechanisms may significantly influence metabolic control in HypoPT and eventually skeletal and extra-skeletal manifestation of the disease. Finally, future data will clarify how the currently available parent vitamin D compounds (ergocalciferol, cholecalciferol, calcifediol) would perform in addressing these specific issues.

Topics: Calcifediol; Calcitriol; Cholecalciferol; Ergocalciferols; Humans; Hypoparathyroidism; Parathyroid Hormone; Vitamin D; Vitamin D Deficiency; Vitamins

Oral administration of active vitamin D is used as a standard medication for the treatment of hypoparathyroidism in our country. Although the dose required for the control of serum calcium level might be different dependent on the pathological type of hypoparathyroidism, the determination of maintenance dose and follow-up are performed by the measurements of serum and urinaly calcium levels. In the future, the clinical use of parathyroid hormone is expected for the treatment of hypoparathyroidism, since it is physiological.

Topics: Calcitriol; Calcium; Drug Therapy, Combination; Ergocalciferols; Humans; Hypoparathyroidism; Teriparatide

Topics: 25-Hydroxyvitamin D 2; Animals; Calcifediol; Calcitriol; Calcium; Cyclic AMP; Ergocalciferols; Glomerular Filtration Rate; Humans; Hyperparathyroidism; Hyperparathyroidism, Secondary; Hypoparathyroidism; Mathematics; Methods; Parathyroid Diseases; Parathyroid Hormone

The effect of 1 alpha OHD2 and 1 alpha OHD3 have been studied in rachitic, normal, and prednisolone-treated male rats. The healing of rickets, the stimulation of intestinal Ca and P transport, the effect on bone mineral, and the induction of renal calcifications have been examined. The two 1-hydroxylated compounds are equally potent in healing rickets, and by comparison with previous data are equivalent to 100-200 IU/micrograms. 1 alpha OHD2 and 1 alpha OHD3 stimulated intestinal Ca and P transport to the same extent and were antagonistic to prednisolone in this respect. 1 alpha OHD2 reduced the number of osteoclasts and increased bone mineral in normal rats in contrast to 1 alpha OHD3, which increased osteoclasts and slightly reduced bone mineral. Prednisolone-induced osteopenia was more effectively counteracted by 1 alpha OHD2 than by 1 alpha OHD3. Mortality rate was higher in rats intoxicated with 1 alpha OHD3 than for rats given 1 alpha OHD2. LD50 was estimated to be five to fifteen times higher for 1 alpha OHD2. Renal calcification were more pronounced after dosing with 1 alpha OHD3 than after treatment with 1 alpha OHD2. When prednisolone was given together with 1 alpha OHD2 or 1 alpha OHD3, renal calcifications were further increased. These observations demonstrate physiological dissimilarities between vitamin D2 and vitamin D3 in rats which are in accordance with a different metabolism of the two vitamins. The findings, in particular that 1 alpha OHD2 is less toxic than 1 alpha OHD3, are of potential clinical importance.

Topics: Animals; Biological Transport; Bone and Bones; Calcium; Chronic Kidney Disease-Mineral and Bone Disorder; Ergocalciferols; Humans; Hydroxycholecalciferols; Hypoparathyroidism; Intestinal Absorption; Intestines; Kidney; Liver; Osteomalacia; Osteoporosis; Phosphorus; Prednisolone; Rats; Rickets; Skin; Vitamin D

The term vitamin D is generally used to describe a number of chemically related compounds with common antirachitic properties, but which have differences in the rapidity of their action, the way they are produced in the body, and the conditions under which their results are optimal. Ergocalciferol, cholecalciferol, 25-hydroxycholecalciferol (calcifediol), dihydrotachysterol, 1 alpha-hydroxycholecalciferol (alfacalcidol), and 1,25-dihydroxycholecalciferol (calcitriol) are currently the most commonly used vitamin D metabolites. In man, cholecalciferol produced on the skin and the fraction obtained from the diet in the gastrointestinal tract are converted in the liver to 25-hydroxycholecalciferol and then in the kidney to 1,25-dihydroxycholecalciferol. The demonstration of these metabolic pathways has helped to elucidate the aetiology of such conditions a hepatobiliary osteodystrophy, drug-induced anticonvulsant osteomalacia, the hypocalcaemia of hypoparathyroidism and above all azotaemic osteodystrophy. In the therapy of azotaemic osteodystrophy, the period of 'vitamin D resistance' when large doses of vitamin D2 and D3 had to be used is now over, and these patients can be efficiently and successfully treated with almost physiological doses of 1 alpha-hydroxycholecalciferol and 1,25-dihydroxycholecalciferol. Attention to diet, calcium supplements and oral phosphate binders are also important. During repetitive haemodialysis, the above principles still hold true, but in some of these patients an osteomalacic syndrome resistant to 1,25-dihydroxycholecalciferol has been recognised. These patients readily become hypercalcaemic when given 1,25-dihydroxycholecalciferol and their fractures and osteomalacia do not improve. Aluminium intoxication, possibly related to the use of impure dialysis fluid, is currently thought to be the most likely explanation of this dialysis osteomalacic syndrome.

Topics: Bone Diseases; Cholecalciferol; Ergocalciferols; Humans; Hypoparathyroidism; Vitamin D

A rapidly growing understanding of the biochemical and physiological processes that underlie the metabolism of vitamin D has provided new insights into the pathogenesis of oestomalacia. Many of the vitamin D--resistant osteomalacia syndromes can now be explained on the basis of defects in the metabolic conversion of vitamin D to the biologically active dihydroxylated metabolite 1,25(OH)2D and perhaps, in some instances, to impairement of the actions of 1,25(OH)2D on target tissues. The availability of this new information has made possible the synthesis of 1-hydroxylated forms of the vitamin for therapeutic use in states of vitamin D resistance. Although many questions regarding the pathogenesis and most effective approaches in the management of osteomalacia remain unanswered, considerable progress has been made in this direction as a result of continued research on the subject.

Topics: Bone Neoplasms; Chemical Phenomena; Chemistry; Cholecalciferol; Dihydroxycholecalciferols; Ergocalciferols; Giant Cell Tumors; Humans; Hydroxycholecalciferols; Hypoparathyroidism; Hypophosphatemia, Familial; Kidney Failure, Chronic; Metabolism, Inborn Errors; Nephrectomy; Osteomalacia; Phosphates; Pseudohypoparathyroidism; Vitamin D; Vitamin D Deficiency

Intestinal malabsorption of calcium and the development of osteomalacia in conservatively treated renal failure is explained by a quantitative deficiency of 1,25-dihydroxycholecalciferol, which also contributes to the development of hypocalcaemia. Excess of 25-hydroxycholecalciferol can substitute for this deficiency. The presence and healing of azotaemic osteomalacia is unrelated to the prevailing plasma [Ca] x [P] product. The data suggest that "vitamin D" acts directly on bone mineralisation, but the claim that this apparent effect is normally due to 25-hydroxycholecalciferol is considered unproven. Most of the phenomena of azotaemic osteodystrophy are encountered in simple vitamin D deficiency; as in that condition, deficiency of 1,25-dihydroxycholecalciferol may be of primary significance in causing secondary hyperparathyroidism in renal failure.

Topics: Bone Resorption; Calcium; Dihydroxycholecalciferols; Drug Resistance; Ergocalciferols; Humans; Hypoparathyroidism; Kidney Failure, Chronic; Phosphates; Pseudohypoparathyroidism; Uremia; Vitamin D

Topics: Anticonvulsants; Bone and Bones; Bone Diseases; Calcium; Cell Membrane Permeability; Cholecalciferol; Cholesterol; Ergocalciferols; Gastrointestinal Diseases; Humans; Hypoparathyroidism; Hypophosphatemia, Familial; Intestinal Mucosa; Kidney; Kidney Failure, Chronic; Parathyroid Hormone; Sarcoidosis; Skin; Vitamin D

Topics: Adenoma; Alkaline Phosphatase; Calcitonin; Calcium; Dihydrotachysterol; Diuretics; Ergocalciferols; Female; Homeostasis; Humans; Hydroxyproline; Hyperparathyroidism; Hypocalcemia; Hypoparathyroidism; Male; Middle Aged; Osteitis Deformans; Parathyroid Glands; Parathyroid Hormone; Parathyroid Neoplasms; Phosphates; Sulfonamides; Thiadiazines

Topics: Adult; Age Factors; Anemia, Pernicious; Calcium; Candidiasis; Child; Dihydrotachysterol; Endocrine System Diseases; Ergocalciferols; Humans; Hypoparathyroidism; Infant, Newborn; Infant, Newborn, Diseases

Topics: Acetates; Calcium; Calcium Chloride; Calcium Phosphates; Cholecalciferol; Citrates; Dihydrotachysterol; Ergocalciferols; Gluconates; Humans; Hypoparathyroidism; Lactates; Magnesium; Parathyroid Hormone; Potassium; Salicylates; Thiosulfates; Vitamin D

Topics: Calcium; Calcium, Dietary; Ergocalciferols; Gluconates; Humans; Hypocalcemia; Hypoparathyroidism; Postoperative Complications; Postoperative Period; Tetany

In order to clarify the mechanisms of thiazide diuretic-induced hypocalciuria, the effect of a thiazide was studied for 7 days in seven patients with hypoparathyroidism on Vitamin D and one on calcium infusion, and seven euparathyroid patients with hypercalciuria. In the control group, calcium excretion (mg/24 hr) fell by 44% from 415 to 232 within 4 days and remained at this level. Plasma total calcium corrected for total protein did not change. In the hypoparathyroid group, calcium excretion fell by 11% from 351 to 311 and then returned to the base line level. Plasma total calcium (mg/100 ml) increased from 10.09 to 10.88, 11.29 and 10.77 at the end of the 2nd, 4th, and 7th day of thiazide administration. In the patient having i.v. calcium and no Vitamin D, neither plasma nor urinary calcium changed significantly. In both groups sodium excretion increased on the first 2 days and fell to or below base line level thereafter. Urinary phosphate, magnesium, and potassium increased, plasma phosphate rose, and magnesium and potassium fell. It is concluded that: (a) The hypocalciuric effect of thiazides requires the presence of parathyroid hormone and is not solely a result of sodium depletion. (b) The hypercalcemic effect of thiazides in hypoparathyroidism is due to increased release of calcium from bone and requires the presence of a pharmacologic dose of Vitamin D. (c) Thiazides enhane the action of parathyroid hormone on bone and kidney; Vitamin D can replace parathyroid hormone in this interaction in bone but not in kidney.

Topics: Adult; Aged; Bone and Bones; Calcium; Chlorothiazide; Cholecalciferol; Clinical Trials as Topic; Dihydrotachysterol; Drug Interactions; Ergocalciferols; Female; Humans; Hypoparathyroidism; Kidney Tubules; Magnesium; Male; Methyclothiazide; Middle Aged; Natriuresis; Parathyroid Hormone; Phosphates; Potassium; Urinary Calculi; Vitamin D

Studies were undertaken in man to evaluate the roles of volume depletion and of the parathyroid glands in mediating the changes in serum and urinary calcium which follow the administration of hydrochlorothiazide, 100 mg twice daily, for 4 days, 42 studies were carried out in 16 normal subjects, 9 patients with hyperparathyroidism, and 7 vitamin D-treated subjects with hypoparathyroidism. In six studies in normal subjects, daily sodium losses during thiazide administration were quantitatively replaced, and in six other studies the effect of equivalent sodium losses produced by furosemide was evaluated. Although the magnitude of sodium losses was similar in three groups during therapy with thiazides, urinary calcium fell and urinary phosphorus increased significantly only in normal subjects and those with hyperparathyroidism; no change occurred in patients with hypoparathyroidism. With the replacement of the thiazide-induced sodium losses by NaCl in normals, urinary calcium did not change as urinary sodium increased 4- to 5-fold. Furosemide administration produced similar sodium losses while urinary calcium remained at or above control levels. After correction for changes in plasma protein concentration caused by thiazide-induced hemoconcentration, mean levels of serum calcium were significantly increased only in subjects with hyperparathyroidism and vitamin D-treated patients with hypoparathyroidism. The results indicate that both depletion of extracellular fluid volume and the presence of the parathyroid glands are necessary for the decrease in urinary calcium in response to thiazide therapy. Both the reduction in urinary calcium and increase in urinary phosphate after the use of thiazides may be due, in part, to potentiation of the action of the parathyroid hormone on the nephron. The rise in serum calcium could be due to thiazide-induced release of calcium from bone into extracellular fluid, particularly in states where bone resorption may be augmented, i.e., vitamin D therapy or hyperparathyroidism.

Topics: Adult; Aged; Blood Proteins; Bone Resorption; Calcium; Clinical Trials as Topic; Ergocalciferols; Extracellular Space; Furosemide; Humans; Hydrochlorothiazide; Hyperparathyroidism; Hypoparathyroidism; Male; Middle Aged; Natriuresis; Phosphates

Topics: Adult; Bone Diseases; Bone Resorption; Calcium; Clinical Trials as Topic; Ergocalciferols; Female; Humans; Hydroxyproline; Hypoparathyroidism; Male; Middle Aged; Phosphates; Vitamin D

Topics: Adolescent; Adult; Alkaline Phosphatase; Calcium; Capsules; Child; Child, Preschool; Clinical Trials as Topic; Drug Hypersensitivity; Ergocalciferols; Female; Humans; Hypoparathyroidism; Male; Middle Aged; Phosphates; Rickets; Tablets

Topics: Calcium; Clinical Trials as Topic; Edetic Acid; Ergocalciferols; Humans; Hypoparathyroidism; Placebos; Thyroidectomy

Calcium and vitamin D treatment does not improve reduced quality of life (QOL) in hypoparathyroidism. Recombinant human (rh) PTH(1-84) therapy improves QOL metrics for up to 5 years. Data on QOL beyond this time point are not available.. To evaluate the effects of 8 years of rhPTH(1-84) therapy on QOL and factors associated with long-term benefit.. Prospective, open-label trial.. Referral center.. Twenty patients with hypoparathyoidism.. RAND 36-Item Short Form Health Survey (SF-36).. rhPTH therapy led to substantial improvement in five of the eight SF-36 domains [vitality, social functioning (SF), mental health (MH), bodily pain (BP) and general health] and three of these domains (SF, MH, BP) were no longer lower than the reference population. The improvement in the mental component summary (MCS) score was sustained through 8 years, while the physical component summary (PCS) score improved through 6 years. A lower baseline QOL score was associated with greater improvement. A threshold value <238 (MCS) and <245 (PCS) predicted long-term improvement in 90% and 100% of the cohort, respectively. In patients whose calcium supplementation was reduced, MCS and PCS scores improved more than those whose supplementation did not decline to the same extent. Improvement in PCS was greater in patients whose calcitriol dosage was reduced and duration of disease was shorter.. rhPTH(1-84) improves long-term well-being in hypoparathyroidism. The improvements are most prominent in those with impaired SF-36 at baseline and those whose requirements for conventional therapy decreased substantially.

Topics: Adult; Aged; Calcitriol; Calcium; Calcium-Regulating Hormones and Agents; Cholecalciferol; Ergocalciferols; Female; Humans; Hypoparathyroidism; Longitudinal Studies; Male; Middle Aged; Parathyroid Hormone; Prospective Studies; Quality of Life; Recombinant Proteins; Treatment Outcome; Vitamin D

Acute onset of quadriplegia is a rare phenomenon seen with hypocalcemia due to hypoparathyroidism. We describe a 33-year gentleman who presented with weakness of all four limbs and areflexia. ECG showed QT abnormality. Nerve conduction study revealed normal sensory and significantly low motor CMAP amplitudes in both the upper and lower extremities. This nerve conduction study can be seen in acute motor neuropathy of various etiologies, among which GBS is the most worrisome. Our patient found to have low calcium and parathyroid hormone level. His symptoms improved after calcium replacement. Occurrence of quadriplegia in hypoparathyroidism, and its improvement after correction of calcium, suggests crucial role of calcium in neuromuscular transmission. One should suspect electrolyte imbalance, like hypocalcemia in patients presenting with nerve conduction features of AMAN variant of GBS.

Topics: Adult; Calcium Gluconate; Ergocalciferols; Humans; Hypocalcemia; Hypoparathyroidism; Male; Neural Conduction; Peripheral Nerves; Quadriplegia

Options for chronic treatment of hypoparathyroidism include calcitriol, recombinant human parathyroid hormone, and high-dose vitamin D (D2). D2 is used in a minority of patients because of fear of prolonged hypercalcemia and renal toxicity. There is a paucity of recent data about D2 use in hypoparathyroidism.. Compare renal function, hypercalcemia, and hypocalcemia in patients with hypoparathyroidism treated chronically with either D2 (D2 group) or calcitriol.. A retrospective study of patients with hypoparathyroidism treated at the University of Maryland Hospital. Participants were identified by a billing record search with diagnosis confirmed by chart review. Thirty patients were identified; 16 were treated chronically with D2, 14 with calcitriol. Data were extracted from medical records.. Serum creatinine and calcium, hospitalizations, and emergency department (ED) visits for hypercalcemia and hypocalcemia.. D2 and calcitriol groups were similar in age (58.9 ± 16.7 vs 50.9 ± 22.6 years, P = 0.28), sex, and treatment duration (17.8 ± 14.2 vs 8.5 ± 4.4 years, P = 0.076). Hospitalization or ED visits for hypocalcemia occurred in none of the D2 group vs four of 14 in the calcitriol group (P = 0.03); three in the calcitriol group had multiple ED visits. There were no differences between D2 and calcitriol groups in hospitalizations or ED visits for hypercalcemia, serum creatinine or calcium, or kidney stones.. We found less morbidity from hypocalcemia in hypoparathyroid patients treated chronically with D2 compared with calcitriol and found no difference in renal function or morbidity from hypercalcemia. Treatment with D2 should be considered in patients with hypoparathyroidism, particularly in those who experience recurrent hypocalcemia.

Topics: Adult; Aged; Calcitriol; Calcium; Creatinine; Emergency Service, Hospital; Ergocalciferols; Female; Hospitalization; Humans; Hypercalcemia; Hypocalcemia; Hypoparathyroidism; Kidney Calculi; Male; Middle Aged; Nephrocalcinosis; Renal Insufficiency; Retrospective Studies; Vitamins

Ipilimumab (a monoclonal antibody against CTLA-4) and nivolumab (a humanized antibody against PD-1) target these immune checkpoint pathways and are used for treatment of melanoma and an increasing number of other cancers. However, they may cause immune-related adverse effects (IRAEs). Although many endocrinopathies are known to be IRAEs, primary hypoparathyroidism with severe hypocalcemia has never been reported. This is the first case of hypoparathyroidism as an IRAE presenting to an Emergency Department with acute hypocalcemia.. A 73-year-old man with metastatic melanoma presented to the Emergency Department for the chief complaints of imbalance, general muscle weakness, abdominal pain and tingling in extremities. He had wide spread metastasis, and begun immunotherapy with concurrent ipilimumab and nivolumab 1.5months ago. At presentation, he had ataxia, paresthesia in the hands and feet, and abdominal cramping. Magnetic resonance imaging of the brain was unremarkable. He was found to be hypocalcemic with undetectable plasma parathyroid hormone. He was admitted for treatment of symptomatic hypocalcemia and was diagnosed with primary hypoparathyroidism. Shortly afterwards, he had thyrotoxicosis manifesting as tachycardia and anxiety, followed by development of primary hypothyroidism. At 4months after the Emergency Department visit, his parathyroid function and thyroid function had not recovered, and required continued thyroid hormone replacement and calcium and vitamin D treatment for hypocalcemia.. Primary hypoparathyroidism caused by ipilimumab and nivolumab may acute manifest with severe symptomatic hypocalcemia. Emergency care providers should be aware of hypoparathyroidism as a new IRAE in this new era of immuno-oncology.

Topics: Aged; Antibodies, Monoclonal; Calcium Gluconate; Ergocalciferols; Hormone Replacement Therapy; Humans; Hypocalcemia; Hypoparathyroidism; Immunotherapy; Magnesium Sulfate; Male; Melanoma; Treatment Outcome; Vitamins

Topics: Calcitriol; Calcium; Ergocalciferols; Humans; Hypocalcemia; Hypoparathyroidism; Vitamin D

Topics: Calcitriol; Calcium; Ergocalciferols; Humans; Hypocalcemia; Hypoparathyroidism; Vitamin D

Hypoparathyroidism and ankylosing spondylitis are two conditions with distinctive features which allow their differentiation. Hypoparathyroidism can be responsible for clinical and radiological changes resembling those seen in patients with ankylosing spondylitis. We report an exceptional case of a patient with an association between ankylosing spondylitis and a severe idiopathic hypoparathyroidism with difficulties in diagnosis. To our knowledge, this is the first case of such an occurrence.

Topics: 25-Hydroxyvitamin D3 1-alpha-Hydroxylase; Anti-Inflammatory Agents; Calcium Gluconate; Dietary Supplements; Ergocalciferols; Humans; Hypocalcemia; Hypoparathyroidism; Male; Middle Aged; Radiography; Severity of Illness Index; Spondylitis, Ankylosing; Tetany; Treatment Outcome

A 16-year-old boy with transfusion-dependent thalassemia major presented with tetany, numbness, bone pain, short stature and pubertal delay. His height SDS score=-2.6, BMI=22.4, spleen was palpable 5 cm and liver 7 cm below the costal margins. The cardio-vascular examination was normal. Laboratory investigations showed a hemoglobin level (8 g/dL), hypocalcemia, hyperphosphatemia and elevated alkaline phosphatase (ALP) with serum 25-OH D below 3 ng/ml and a normal magnesium level. Serum parathyroid hormone (PTH) level was lower (21 pg/mL; normal 16-70 pg/mL) than expected for the degree of hypocalcemia. Serum ferritin concentration was 4442 ug/L, insulin-like growth factor I (IGF-I) was 31 microg/L (normal 122- 286 microg/L), free T4 was 13.1 microg/dL, TSH 1.2 mIU/ml. These results revealed a combined vitamin D-parathyroid defect. Peak growth hormone (GH) responses to clonidine and glucagon tests were 7.6 ng/ml and 6.2 ng/ml, respectively. Serum LH and FSH concentrations were below 0.5 U/L and testosterone was below 10 ng/dl. Radiographs revealed osteopenia of the phalanges and long bones and DXA scanning revealed low BMD Z-score of the femoral neck and 4th and 5th lumbar spines. MRI showed evidence of hemosiderin deposition in the pituitary. The patient was started on oral daily calcium carbonate (1500 mg elemental calcium) and vitamin D2 (calciferol) 25,000 IU/day and intensive iron chelation therapy. A low dose of IM testosterone enanthate (1 mg/kg/month) was injected for 6 months. Follow-up after 4, 8 and 12 months revealed normal Ca, PO4, ALP, and 25-OH D concentrations and disappearance of spasms and numbness and increased growth velocity. In conclusion, investigating calcium homeostasis at regular intervals and early management of any abnormality can preclude the occurrence of complications.

Topics: Adolescent; beta-Thalassemia; Calcium; Ergocalciferols; Humans; Hypoparathyroidism; Hypopituitarism; Iron Chelating Agents; Male

Topics: Aged; Calcium; Drug Therapy, Combination; Ergocalciferols; Humans; Hypocalcemia; Hypoparathyroidism; Male; Psoriasis

We have examined the role of PTH in the postnatal state in a mouse model of PTH deficiency generated by targeting the Pth gene in embryonic stem cells. Mice homozygous for the ablated allele, when maintained on a normal calcium intake, developed hypocalcemia, hyperphosphatemia, and low circulating 1,25-dihydroxyvitamin D(3) [1,25(OH)(2)D(3)] levels consistent with primary hypoparathyroidism. Bone turnover was reduced, leading to increased trabecular and cortical bone volume in PTH-deficient mice. When mutant mice were placed on a low-calcium diet, renal 25-hydroxyvitamin D 1 alpha-hydroxylase expression increased despite the absence of PTH, leading to a rise in circulating 1,25(OH)(2)D(3) levels, marked osteoclastogenesis, and profound bone resorption. These studies demonstrate the dependence of the skeletal phenotype in animals with genetically depleted PTH on the external environment as well as on internal hormonal and ionic circulatory factors. They also show that, although PTH action is the first defense against hypocalcemia, 1,25(OH)(2)D(3) can be mobilized, even in the absence of PTH, to guard against extreme calcium deficiency.

Topics: 25-Hydroxyvitamin D3 1-alpha-Hydroxylase; Animals; Animals, Newborn; Bone and Bones; Bone Diseases; Bone Remodeling; Bone Resorption; Calcium, Dietary; Cell Division; Dose-Response Relationship, Drug; Ergocalciferols; Homeostasis; Homozygote; Hypoparathyroidism; Kidney; Mice; Mice, Knockout; Osteoclasts; Parathyroid Glands; Parathyroid Hormone

Fahr's disease associates various degrees of neuropsychological impairment and calcium deposits in the basal ganglia. We report 3 cases. The first case was a 54-year-old man with hemichorea of one-year duration. Laboratory results demonstrated idiopathic hypoparathyroidism. In the second case, a 23-year-old man treated for epilepsia for 8 years was hospitalized for subintrant episodes and hemichorea. Dysmorphism and laboratory results led to the diagnosis of pseudo-hypothyroidism. The third case was a 62-year-old woman with generalized seizures of epilepsia and dementia of two-month duration. Physical examination revealed extra-pyramidal rigidity. Hyperparathyroidism due to an adenoma was confirmed histologically. In all three patients, correction of phosphocalcium levels led to clinical improvement, particularly with disappearance of the epileptic seizures and abnormal movements. Clinical expression of Fahr's syndrome varies greatly. Symptoms include psychiatric disorders, epileptic seizures, extra-pyramidal syndrome and various neurological conditions. Diagnosis requires CT brain scan which identifies calcium deposits in the basal ganglia. The main cause is hypoparathyroidism, whether primary or post-operative. Cases due to other causes of dysparathyroidism are rare. The pathophysiology of this condition remains unknown and results of treatment are often unsatisfactory. Since correcting the impaired calcium phosphorus metabolism often leads to considerable improvement, it is essential to systematically search for dysparathyroidism in patients presenting with neuropsychologic manifestations associated with calcifications of the basal ganglia.

Topics: Adult; Athetosis; Basal Ganglia Diseases; Calcinosis; Calcium; Chorea; Drug Therapy, Combination; Epilepsy, Tonic-Clonic; Ergocalciferols; Female; Humans; Hyperparathyroidism; Hypoparathyroidism; Male; Middle Aged; Syndrome; Tomography, X-Ray Computed

Parathyroid autotransplantation was first described in 1907 by Halsted. However, this simple and effective method of preserving parathyroid function has been used with increasing frequency only during the past 25 years. Beginning in the late 1960s, our group has transplanted normal parathyroid tissue into the ipsilateral sternocleidomastoid muscle whenever these glands could not be preserved in situ with adequate blood supply. In addition, if the blood supply of all four parathyroid glands appeared compromised, cryopreservation of parathyroid tissue was performed in case the autotransplanted tissue did not function after surgery. Since 1970, 393 patients underwent a total thyroidectomy. Parathyroid glands that could not be saved in situ were biopsied to confirm their identity by frozen section and then autotransplanted. Of the 393 patients who underwent a total thyroidectomy, 261 patients required transplantation of one or more glands. Among those 261 patients who underwent selective parathyroid autotransplantation, 33 (13%) required temporary calcium and vitamin D supplementation. Of these 33 patients, 2 (less than 1%) had permanent hypoparathyroidism and are receiving long-term vitamin D therapy.

Topics: Adolescent; Adult; Aged; Aged, 80 and over; Calcium Carbonate; Child; Child, Preschool; Dihydrotachysterol; Ergocalciferols; Follow-Up Studies; Humans; Hypocalcemia; Hypoparathyroidism; Middle Aged; Neck Muscles; Parathyroid Glands; Phosphates; Thyroidectomy; Transplantation, Autologous

A 76-year-old female patient who had been taking vitamin D2 100,000 U/day for more than 14 years due to hypoparathyroidism following total throidectomy was admitted because of protracted hypercalcemia. On admission, the levels of serum vitamin D2 (99.8 ng/ml) and 25-OHD2 (356 ng/ml) were very high, and 1,25-(OH)2D2 was low (4.0-18.7 pg/ml). Serum D3' 25-OHD3 and 1,25-(OH)2D3 were below the normal range. Despite intensive hydration with saline, intravenous hyperalimentation with phosphate- and calcium-free nutrients, and administration of glucocorticoid and calcitonin, the hypercalcemia persisted, accompanied by hypoproteinemia, edema, pleural effusion and congestive heart failure. The serum D2 and 25-OHD2 concentrations remained high and were accompanied by a gradual increase in 1,25-(OH)2D2 (121 pg/ml), which further increased after the administration of bisphosphonate (pamidronate) to 183 pg/ml. Seventeen months later, serum calcium and 1,25-(OH)2D2 were normalized but serum D2 and 25-OHD2 remained high. The serum 24,25-(OH)2D2/25-OHD2 ratio was relatively constant throughout her clinical course, whereas the low serum 1,25-(OH)2D2/25-OHD2 ratio at admission gradually increased during admission, suggesting that the increase in serum 1,25-(OH)2D2 is due to increased production rather than decreased degradation. The administration of pamidronate further increased serum 1,25-(OH)2D2. These features of the clinical course demonstrate that the 1,25-dihydroxyvitamin D concentration in hypercalcemic patients with protracted vitamin D intoxication may be decreased, normal or increased. Possible factors responsible for a protracted increase in serum 1,25-(OH)2D2 are body weight loss, hypoproteinemia, and phosphate depletion. In addition, some bisphosphonates would certainly promote PTH-independent production of 1,25-(OH)2D2.

Topics: Aged; Ergocalciferols; Female; Humans; Hypercalcemia; Hypoparathyroidism

Topics: Adult; Calcinosis; Cerebrovascular Disorders; Ergocalciferols; Female; Humans; Hypoparathyroidism; Skull; Syndrome; Tomography, X-Ray Computed

Topics: Basal Ganglia Diseases; Calcinosis; Child; Ergocalciferols; Humans; Hypoparathyroidism; Male; Spasm; Tomography, X-Ray Computed

The response of circulating 1,25-dihydroxyvitamin D [1,25-(OH)2D] to challenge with vitamin D treatment both before and after 7-10 days of prednisone therapy (25 mg/day) was investigated in five anephric subjects, six patients with chronic renal failure (CRF), two patients with vitamin D intoxication and four patients with hypoparathyroidism. In anephric subjects serum 25-hydroxyvitamin D [25-(OH)D] rose from 58 +/- 48 (SD) to 377 +/- 221 (SD) nmol/l after administration of 150 micrograms of 25-(OH)D3 for 1 month. Serum 1,25-(OH)2D, which was barely detectable in only two out of five patients under basal conditions, rose to 30 +/- 21 pmol/l after 2 weeks of therapy with 25-(OH)D3, but fell to 10 +/- 5 pmol/l during prednisone treatment. In CRF patients circulating 1,25-(OH)2D rose from 37 +/- 24 to 58 +/- 24 pmol/l during 25-(OH)D3 therapy, but fell to 41 +/- 31 pmol/l during prednisone treatment. In two patients with rheumatoid arthritis, hypercalcaemia due to vitamin D intoxication was associated with raised levels of 1,25-(OH)2D (288 and 317 pmol/l). Administration of prednisone resulted in suppression of 1,25-(OH)2D levels (132 and 96 pmol/l respectively) and reduction of serum calcium to within the normal range. In the hypoparathyroid patients prednisone therapy did not affect circulating 25-(OH)D levels but serum 1,25-(OH)2D fell from 192 +/- 42 to 117 +/- 23 pmol/l and serum calcium from 2.41 +/- 0.21 to 2.20 +/- 0.05 mmol/l.(ABSTRACT TRUNCATED AT 250 WORDS)

Topics: Calcifediol; Calcitriol; Calcium; Ergocalciferols; Female; Humans; Hydroxycholecalciferols; Hypoparathyroidism; Kidney Failure, Chronic; Prednisone; Renal Dialysis; Vitamin D

In 23 patients with hypoparathyroidism or pseudohypoparathyroidism treated with vitamin D, and in whom the dosage was adjusted downward or upward in response to hypercalcemia or hypocalcemia respectively, assays of serum 25-hydroxyvitamin D (25-OHD) were carried out in addition to the usual serum calcium assays. In 120 assays there was a significant correlation between serum 25-OHD levels and serum calcium levels (corrected for serum albumin). There was, however, no clear distinction between the 25-OHD levels of patients who were hypocalcemic, normocalcemic or hypercalcemic. The highest serum 25-OHD level found in a hypocalcemic patient was 1193 nmol/L and the lowest serum 25-OHD level found in a hypercalcemic patient was 605 nmol/L. It was not possible to predict subsequent episodes of hypocalcemia or hypercalcemia from the serum 25-OHD levels. The 25-OHD assay was found to be useful only in checking compliance. We conclude that the assay of serum 25-OHD is of no more value than serum calcium alone in the management of compliant patients.

Topics: 25-Hydroxyvitamin D 2; Adult; Aged; Calcium; Ergocalciferols; Female; Humans; Hypoparathyroidism; Male; Middle Aged; Pseudohypoparathyroidism

Topics: Adult; Calcium; Ergocalciferols; Female; Follow-Up Studies; Humans; Hypoparathyroidism; Male; Middle Aged; Time Factors

This interlaboratory study on determination of 25-hydroxyvitamin D (25-OH-D) in serum involved 15 laboratories in eight European countries. All could distinguish between normal (50 +/- 31 nmol/L, mean +/- SD) and grossly increased concentrations, but for eight laboratories the results for serum samples with low and normal 25-OH-D content overlapped. In general, values were well reproducible, but interlaboratory variation in 25-OH-D measurement was large, 24,25(OH)2D3 interfering in most of the assays. We present evidence in favor of chromatography before assay, as opposed to nonchromatographic methods. Liquid chromatography with ultraviolet detection for quantifying 25-OH-D2 and 25-OH-D3 appears to be an appropriate reference method, whereas competitive protein binding assay is the method of choice for routine determinations. Control sera with subnormal, normal, and above-normal concentrations of 25-OH-D3 are needed for use in standardization of 25-OH-D assays.

Topics: 25-Hydroxyvitamin D 2; Calcifediol; Carrier Proteins; Charcoal; Chemistry, Clinical; Cholecalciferol; Chromatography, High Pressure Liquid; Ergocalciferols; Europe; False Positive Reactions; Humans; Hypoparathyroidism; Spectrophotometry, Ultraviolet; Vitamin D; Vitamin D-Binding Protein

Topics: Adult; Breast Feeding; Ergocalciferols; Female; Humans; Hypoparathyroidism; Infant, Newborn; Milk, Human; Pregnancy; Pregnancy Complications

Topics: Adult; Calcium; Ergocalciferols; Humans; Hypocalcemia; Hypoparathyroidism; Male; Sarcoidosis

A sensitive radioimmunoassay for 1,25-dihydroxy vitamin D2 was developed using a sheep antiserum which preferentially reacts with 1-hydroxylated forms of vitamin D. An improved isolation procedure was also developed using acetonitrile for the initial extraction of serum followed by chromatography on cartridges of C18 silica and high pressure liquid chromatography eluted with a ternary solvent system to separate 1,25-dihydroxy vitamin D2 and 1,25-dihydroxy vitamin D3. 25-hydroxy vitamin D2 and 25-hydroxy vitamin D3 were separated by further reverse phase high pressure liquid chromatography prior to competitive protein binding assay. The limits of detection were 4.3 pmol/1 (2.0 pg/ml) for the 1,25-dihydroxy metabolites and 1.25 nmol/1 (0.5 ng/ml) for both 25-hydroxy vitamin D2 and 25-hydroxy vitamin D3. 25-hydroxy vitamin D2 ranged from 2.0 to 11.3 nmol/1 (0.8-4.5 ng/ml) with a mean of 4.75 nmol/1 (1.9 ng/ml) in thirteen healthy British adults and this accounted for 9.0% of the mean total 25-hydroxy vitamin D. 1,25-dihydroxy vitamin D2 was detected in the sera of only one of these subjects whereas 1,25-dihydroxy vitamin D3 was present in all ranging from 48 to 163 pmol/1 (20-65 pg/ml) with a mean of 100 pmol/1 (42 pg/ml). Both 1,25-dihydroxy vitamin D2 and 1,25-dihydroxy vitamin D3 were detected in the sera of hypoparathyroid patients treated with vitamin D2 but the relationship between 25-hydroxy vitamin D and 1,25-dihydroxy vitamin D was complex. For example, when an excess of 25-hydroxy vitamin D2 was present the serum concentration of 1,25-dihydroxy vitamin D3 was disproportionately high. Conversely, in patients who had previously been treated with vitamin D2 but were receiving only vitamin D3 at the time of study, the major 25-hydroxy metabolite was in the vitamin D3 form and there was a disproportionately high amount of 1,25-dihydroxy vitamin D2. Total 1,25-dihydroxy vitamin D ranged from 110 to 400 pmol/1 (45-165 pg/ml) and was above the upper limit of normal for 1,25-dihydroxy vitamin D3 in half of these hypoparathyroid patients treated with pharmacological doses of vitamin D.

Topics: 25-Hydroxyvitamin D 2; Calcifediol; Calcitriol; Chromatography, High Pressure Liquid; Ergocalciferols; Humans; Hydroxylation; Hypoparathyroidism; Radioimmunoassay

A 17-year-old girl with thalassemia major experienced tetany. The serum calcium level was 5.5 mg/dL, and the phosphorus level was 6.3 mg/dL. Serum levels of parathyroid hormone (PTH) and 25-hydroxyvitamin D (25-OHD) were subnormal at 125 pg/mL and 8.1 ng/mL, respectively, As a result of these findings, serum 25-OHD and PTH levels were measured in an additional 12 patients with thalassemia major. Low levels of both 25-OHD and PTH were found frequently. An increase in serum 25-OHD levels was noted in each of four patients who were examined after iron chelation therapy.

Topics: 25-Hydroxyvitamin D 2; Adolescent; Adult; Child; Deferoxamine; Ergocalciferols; Female; Humans; Hypoparathyroidism; Male; Thalassemia

Serum calcium, phosphorus, calcitonin, parathyroid hormone, 25-hydroxyvitamin D (25OHD), and 1,25-dihydroxyvitamin D [1,25-(OH)2D] were measured in 6 women and 2 men with medullary carcinoma of the thyroid, 22 normal subjects, 5 patients with chronic renal failure, and 5 patients with primary hyperparathyroidism. Serum 1,25-(OH)2D levels were significantly higher in patients with primary hyperparathyroidism and lower in patients with chronic renal failure than in normal subjects. In patients with medullary carcinoma of the thyroid, the serum calcitonin levels were elevated, but the parathyroid hormone and 1,25-(OH)2D levels were within normal ranges. The serum 25OHD levels were not significantly different in any group. It is concluded that chronic elevation of serum calcitonin has no effect on the serum 1,25-(OH)2D level.

Topics: 25-Hydroxyvitamin D 2; Adolescent; Adult; Calcitonin; Calcitriol; Carcinoma; Ergocalciferols; Female; Humans; Hypoparathyroidism; Kidney Failure, Chronic; Male; Middle Aged; Thyroid Neoplasms

Fourteen patients with pseudohypoparathyroidism, 17 with idiopathic hypoparathyroidism, and 12 with postoperative hypoparathyroidism were treated with vitamin D2, dihydrotachysterol, 1 alpha-hydroxyvitamin D3)1 alpha-OHD3), and 1,25-dihydroxyvitamin D3 for 6-18 months. The optimal maintenance dose or minimum daily dose of 1,25-dihydroxyvitamin D3 to maintain serum calcium at approximately 8.5 mg/100 ml and control all the clinical symptoms was 1.3 +/- 0.16 micrograms/day (mean +/- SE) in pseudohypoparathyroidism, 1.5 +/- 0.18 micrograms/day in idiopathic hypoparathyroidism, and 1.9 +/- 0.50 micrograms/day in postoperative hypoparathyroidism. There was no significant difference in the optimal maintenance dose among the 3 groups. The optimal maintenance dose of 1 alpha-OHD3, however, was 2.0 +/- 0.12 micrograms/day in pseudohypoparathyroidism, significantly lower than that in idiopathic hypoparathyroidism (3.5 +/-0.29 micrograms/day; P less than 0.001) and in postoperative hypoparathyroidism (4.89 +/- 0.54 micrograms/day; P less than 0.001). Significantly lower doses were required in the treatment of idiopathic hypoparathyroidism than in postoperative hypoparathyroidism (P less than 0.05). No significant difference was found in the optimal maintenance dose of dihydrotachysterol and vitamin D2 among the 3 groups. The average pretreatment serum calcium levels and clinical manifestations were indistinguishable among the 3 groups of patients. This suggests that such a difference in the optimal maintenance dose of 1 alpha-OHD3 is ascribed not to the difference in the severity of hypoparathyroidism, but most probably to differences in the pathophysiological processes in pseudohypoparathyroidism and idiopathic or postoperative hypoparathyroidism. The excess parathyroid hormone levels in blood of patients with pseudohypoparathyroidism (and not in other types of hypoparathyroidism) may explain such a difference.

Topics: Adolescent; Adult; Aged; Body Weight; Calcifediol; Calcitriol; Calcium; Dihydrotachysterol; Ergocalciferols; Female; Humans; Hydroxycholecalciferols; Hypoparathyroidism; Male; Middle Aged; Parathyroid Hormone; Phosphorus; Postoperative Complications; Pseudohypoparathyroidism; Vitamin D

The basal values of plasma vitamin D metabolites were evaluated in patients with primary hyperparathyroidism (1 degree HPT, n = 31), hypoparathyroidism (HP, n = 7), pseudohypoparathyroidism (PHP, n = 4) and normal controls (n = 21). Plasma 25-hydroxyvitamin D (25-OH-D) in 1 degree HPT (9.0 +/0 7.3 ng/ml, mean SD) was significantly lower than that of normal controls (17.9 +/- 5.5ng/ml)(p less than 0.001), and in particular 1 degree HPT classified as the skeletal type showed extremely low value (4.7 +/- 4.6 ng/ml). Plasma 1, 25-dihydroxyvitamin D [1, 25-(OH)2D] was significantly higher in 1 degree HPT (69.1 +/- 31.4pg/ml)(p less than 0.001) and significantly lower in Hp (15.2 +/- 11.0 pg/ml) (p less than 0.001) compared to normal controls (37.2 +/- 13.8pg/ml), although there was no significant difference in PHP (22.3 +/- 17.5 pg/ml). Plasma 24, 25-dihydroxyvitamin D [24,, 25-(OH)2D] in 1 degree HPT (1.06 +/- 0.55 ng/ml) was significantly lower than that of normal controls (1.73 +/- 0.62 ng/ml) (p less than 0.05), and particularly 1 degree HPT classified as the skeletal type showed a marked low value (0.85 +/- 0.27 ng/ml), whereas no significant differences were seen in HP (1.84 +/- 0.46 ng/ml) or PHP (1.34 +/- 0.22 ng/ml). There were slight but significant correlations between either plasma 25-OH-D and 1, 25-(OH)2D (r = -0.350, p less than 0.05), or plasma 25-OH-D and 24, 25-(OH)2D (r = 0.356, p less than 0.05), or plasma 1, 25-(OH)2D and 24, 25-(OH)2D (r = -0.444, p less than 0.01) in all subjects. In addition, relationships between plasma vitamin D metabolites and other indicators of parathyroid function in all subjects were analyzed. There were positive correlations between plasma 1, 25-(OH)2D and serum Ca (r = -0.621, p less than 0.001) or urinary cAMP (r = -0.671, p less than 0.001) or nephrogenous cAMP (r = -0.689, p less than 0.001), while negative correlations were seen between plasma 1, 25-(OH)2D and serum P (r = -0.680, p less than 0.001) or %TRP (r = -0.663, p less than 0.001). On the other hand, there were negative correlations between plasma 24, 25-(OH)2D and serum Ca (r = -0.457, p less than 0.01) or urinary cAMP (r = -0.562, p less than 0.005) or nephrogenous cAMp (r = -0.561, p less than 0.005), and a positive correlation was seen between plasma 24, 25-(OH)2D and %TRP (r = 0.519, p less than 0.005). After parathyroidectomy, a distinct depression of plasma 1, 25-(OH)2D and reciprocal elevation of plasma 24, 25-(OH)2D were observed in 1 degree

Topics: 25-Hydroxyvitamin D 2; Adolescent; Adult; Aged; Calcium; Dihydroxycholecalciferols; Ergocalciferols; Female; Humans; Hydroxycholecalciferols; Hypoparathyroidism; Male; Middle Aged; Phosphorus; Pseudohypoparathyroidism

Topics: 24,25-Dihydroxyvitamin D 3; Calcifediol; Calcitriol; Calcium; Chronic Kidney Disease-Mineral and Bone Disorder; Dihydroxycholecalciferols; Ergocalciferols; Humans; Hydroxycholecalciferols; Hypoparathyroidism; Intestinal Absorption; Osteomalacia; Osteoporosis; Rickets; Tetany; Vitamin D

Oral administration of prednisone (30 mg/day for 9 days) to six normal individuals induced a significant rise in the concentration of serum 1,25-dihydroxyvitamin D [1,25-(OH)2D] within 2 days. In four patients with primary hyperparathyroidism a larger increase of 1,25-(OH)-2D was observed within 3 days. In these patients the 1,25-(OH)-2D concentration remained elevated during the whole period of prednisone administration (10 days) whereas in the control group it had returned to basal levels or below after 9 days of prednisone administration. This response appeared dependent upon parathyroid hormone (PTH) as we found no change in the (basally low) 1,25-(OH)2D concentrations in five patients with hypoparathyroidism during 3-4 days of prednisone administration (30 mg/day). In these patients vitamin D medication had been interrupted 3-5 days before the administration of prednisone, whereafter serum calcium was kept between 2.10 and 2.30 mmol/1 by means of calcium infusion. The response of 1,25-(OH)2D to prednisone is best explained by a stimulatory action of glucocorticoids upon PTH secretion or by the induction of increased PTH sensitivity.

Topics: 24,25-Dihydroxyvitamin D 3; 25-Hydroxyvitamin D 2; Adult; Aged; Calcitriol; Dihydroxycholecalciferols; Ergocalciferols; Female; Humans; Hyperparathyroidism; Hypoparathyroidism; Male; Middle Aged; Prednisone

Topics: Animals; Calcium; Cat Diseases; Cats; Dihydrotachysterol; Dihydroxycholecalciferols; Dog Diseases; Dogs; Ergocalciferols; Hypoparathyroidism; Vitamin D

Topics: Calcium Metabolism Disorders; Cholecalciferol; Chronic Kidney Disease-Mineral and Bone Disorder; Ergocalciferols; Humans; Hypoparathyroidism; Osteomalacia; Osteoporosis; Phosphorus Metabolism Disorders; Renal Dialysis; Vitamin D

Topics: Calcium; Dihydrotachysterol; Ergocalciferols; Humans; Hypoparathyroidism

Only moderately reduced serum 1,25-dihydroxyvitamin D [1,25(OH)2D3] levels were found in 13 hypoparathyroid patients and in 1 pseudohypoparathyroid patient, indicating that factors other than parathyroid hormone are able to mediate the basal production of 1,25(OH)2D3. A significant correlation was found between the levels of circulating 25-hydroxyvitamin D (25OHD) and 1,25(OH)2D, suggesting that a high concentration of 25OHD was able to increase the renal production of 1,25(OH)2D, whereas hypocalcemia and changes in serum phosphate became less important in the present situation. The serum 25OHD and 1,25(OH)2D concentrations were followed during the change of treatment from ergocalciferol to 1 alpha-hydroxycholecalciferol (1 alpha-OHD3). A biological half-life of 3 weeks could be estimated for the plasma 25OHD. The increase in serum 1,25(OH)2D correlated with the dose of 1 alpha-OHD3 given, whereas no correlation was found between the serum calcium level and the 1,25(OH)2D concentration. Unexpected increases and decreases in serum calcium were observed at the same dose of 1 alpha-OHD3 in the same patient and at serum 1,25(OH)2D concentrations within the physiological range.

Topics: 25-Hydroxyvitamin D 2; Adolescent; Calcitriol; Calcium; Dihydroxycholecalciferols; Ergocalciferols; Humans; Hydroxycholecalciferols; Hypoparathyroidism; Kinetics; Male

Vitamin D and its metabolites 25-hydroxyvitamin D (25OHD), 24,25-dihydroxyvitamin D [24,25-(OH)2D], 25,26-(OH)2D, and 1,25-(OH)2D were measured after separation on high pressure liquid chromatography in sera from two hypoparathyroid patients treated with high doses of vitamin D2. Fractions with displacement activities in the competitive protein-binding assays were found which were not detectable in sera from controls not supplemented with vitamin D2. These fractions, presumably representing vitamin D2 metabolites, were quantitated separately from the vitamin D3 metabolites. After a change of treatment from milligram doses of vitamin D2 to microgram doses of 1 alpha OHD3, the serum metabolites of vitamin D2 and vitamin D3 were followed from 8-13 months. During the first 2-3 months, there was an initial relatively rapid fall in serum vitamin D2 levels and metabolites, followed by a slower decline. High levels of vitamin D2 metabolites were still present after 13 months. Taking into account the marked preponderance of 25OHD2 to 25OHD3, the relative concentration of (OH)2D3 metabolites were higher than expected, which might indicate a preferential 25OHD3 hydroxylation or alternatively, a more rapid degradation of vitamin D2 metabolites in these patients. The high and sustained release of vitamin D2, presumably from fat stores, more than a year after vitamin D2 ingestion was stopped has obvious clinical implications and should be considered in the long term follow-up of patients shifted from the traditional high doses of vitamin D to the newly synthesized 1 alpha OHD3 or 1,25(OH)2D3.

Topics: Adult; Child; Chromatography, High Pressure Liquid; Dihydroxycholecalciferols; Ergocalciferols; Female; Humans; Hydroxycholecalciferols; Hypoparathyroidism; Kinetics; Male; Vitamin D

Topics: Chronic Disease; Ergocalciferols; Female; Humans; Hypocalcemia; Hypoparathyroidism; Male; Renin

In twenty-one patients with chronic hypoparathyroidism a close correlation was observed between the prescribed dose of ergocalciferol and mean serum 25-hydroxycalciferol levels. In the 75 serum specimens examined, a significant correlation was found between 25-hydroxycalciferol levels and calcium concentrations. The measurement of serum 25-hydroxycalciferol levels is of potential clinical value in the prediction of hyper- or hypocalcaemic episodes and in monitoring patient compliance.

Topics: Adolescent; Adult; Aged; Calcium; Ergocalciferols; Female; Humans; Hydroxycholecalciferols; Hypoparathyroidism; Male; Middle Aged

Topics: Adolescent; Adult; Dihydrotachysterol; Ergocalciferols; Female; Humans; Hydroxycholecalciferols; Hypoparathyroidism; Male; Middle Aged; Pseudohypoparathyroidism

25-Hydroxyvitamin D (25-OHD) levels were measured in 39 patients with metabolic bone disease or hypoparathyroidism who had been treated with a constant high dose of vitamin D2 or D3 for at least 12 weeks. Plasma 25-OHD levels rose with increasing dosage, the relationship between dose and plasma level being approximately linear whether or not the dose was expressed on a weight-corrected basis. A therapeutic range of 25-OHD to be expected when patients with these conditions are treated with vitamin D has been established. There may be certain exceptions in which plasma 25-OHD levels within the range are associated with either an inadequate response to treatment or, conversely, the hypercalcaemia of vitamin D toxicity. There was no correlation between plasma calcium level and 25-OHD concentration in the group of patients studied. There was also no difference between the dose/25-OHD relationship of patients treated with vitamin D2 and that of patients receiving vitamin D3. Ten patients were started on treatment with large doses of vitamin D during the period of the study. The rate of rise of plasma 25-OHD was followed during treatment. The incremental rise in 25-OHD was calculated at the end of the first week of treatment in terms of dose per unit body weight. The rate of rise of plasma 25-OHD level was highly correlated with the dose used. Plasma 25-OHD levels after one weeks' treatment were only 15-20% of the expected steady-state level on the same dosage. The importance of a high priming dose when a rapid response is needed is thus emphasised.

Topics: Adolescent; Adult; Aged; Body Weight; Bone Diseases; Child; Child, Preschool; Cholecalciferol; Dose-Response Relationship, Drug; Ergocalciferols; Female; Humans; Hydroxycholecalciferols; Hypercalcemia; Hypoparathyroidism; Infant; Male; Middle Aged; Vitamin D

Topics: Blood Glucose; Calcium; Ergocalciferols; Female; Glucose; Glucose Tolerance Test; Humans; Hypocalcemia; Hypoparathyroidism; Hypophosphatemia, Familial; Insulin; Insulin Secretion; Male; Theophylline

The aims of this study were to determine the dose of vitamin D2 that maintains the serum calcium level within the normal range in hypoparathyroid and pseudohypoparathyroid children and to establish a safe and quickly acting dose for initiating therapy in symptomatic patients. The dose requirement for maintenance therapy was studied in 11 patients and initiation therapy was studied in five newly diagnosed hypocalcemic patients. The results show that (1) the maintenance requirement of vitamin D2 is proportional to body weight and averages 2,000 IU (50 microgram)/kg/day for children of all ages and with all types of hypoparathyroid disorders and the (2) in newly diagnosed symptomatic patients, carefully controlled administration of 8,000 IU (200 microgram) vitamin D2/kg/day for the first one to two weeks corrects hypocalcemia quickly and safely.

Topics: Adolescent; Adult; Body Weight; Calcium; Child; Child, Preschool; Drug Administration Schedule; Ergocalciferols; Female; Humans; Hypocalcemia; Hypoparathyroidism; Male; Pseudohypoparathyroidism; Vitamin D

Topics: Calcium; Child; Diet; Dihydrotachysterol; Ergocalciferols; Humans; Hypercalcemia; Hypocalcemia; Hypoparathyroidism; Magnesium; Male; Phosphorus; Vitamin D

Topics: Aged; Calcium; Cholecalciferol; Ergocalciferols; Esophagus; Female; Humans; Hypercalcemia; Hypoparathyroidism; Laryngectomy; Male; Middle Aged; Parathyroid Hormone; Pharyngectomy; Postoperative Complications; Vitamin D

Topics: Calcium; Ergocalciferols; Female; Humans; Hypercalcemia; Hypoparathyroidism; Hypothyroidism; Middle Aged; Parathyroid Hormone; Prednisone; Thyroid (USP); Vitamin D

Topics: Aged; Calcium; Candidiasis, Cutaneous; Cyanosis; Dermatitis, Exfoliative; Electrocardiography; Ergocalciferols; Female; Humans; Hypoparathyroidism; Laryngismus; Phosphates; Skin Diseases; Thyroid Function Tests; Thyroid Neoplasms; Thyroidectomy; Thyroxine; Tracheotomy

Topics: Adolescent; Anticonvulsants; Body Height; Body Weight; Calcium; Child; Dihydrotachysterol; Epilepsy, Tonic-Clonic; Ergocalciferols; Humans; Hypocalcemia; Hypoparathyroidism; Male; Middle Aged; Osteomalacia; Rickets; Time Factors; Ultraviolet Therapy; Vitamin D

A patient of 29 years is described with Noonan's syndrome and idiopathic hypoparathyroidism, who presented with epilepsy and myoclonus. Correction of the hypocalcaemia resulted in improvement of his myoclonus and psychiatric abnormalities. The embryological significance of the association is discussed.

Topics: Adult; Aluminum Hydroxide; Calcium; Depression; Electroencephalography; Epilepsy; Ergocalciferols; Fluorescein Angiography; Humans; Hypoparathyroidism; Intelligence; Male; Myoclonus; Radiography; Retinal Vessels; Skull; Spinal Canal; Turner Syndrome

Topics: Calcium; Ergocalciferols; Gluconates; Humans; Hypoparathyroidism; Injections, Intramuscular; Male; Middle Aged; Phosphorus; Skin Manifestations

Topics: Acute Kidney Injury; Adult; Ergocalciferols; Female; Humans; Hypercalcemia; Hypoparathyroidism; Kidney Function Tests; Vitamin D

Topics: Anticonvulsants; Asia; Bone Resorption; Calcium; Calcium, Dietary; Epilepsy; Ergocalciferols; Humans; Hydroxycholecalciferols; Hyperparathyroidism; Hypoparathyroidism; Kidney; Kidney Failure, Chronic; Osteomalacia; Pigmentation; Renal Dialysis; Rickets; Scotland; Ultraviolet Rays; Vitamin D

Topics: Adolescent; Calcium; Ergocalciferols; Female; Humans; Hypoparathyroidism; Magnesium; Magnesium Deficiency; Vitamin D

Topics: Adolescent; Adult; Calcium; Child; Electromyography; Ergocalciferols; Female; Humans; Hypoparathyroidism; Male; Muscle Spindles; Muscles; Neuromuscular Junction; Stuttering

Topics: Adenoma; Adult; Alkaline Phosphatase; Calcium; Cardiomegaly; Cardiomyopathies; Ergocalciferols; Female; Humans; Hyperparathyroidism; Hypocalcemia; Hypoparathyroidism; Infant, Newborn; Infant, Newborn, Diseases; Parathyroid Hormone; Parathyroid Neoplasms; Perfusion; Phosphorus; Pregnancy; Pregnancy Complications

Topics: Adenoma; Adult; Aged; Calcium; Carcinoma, Papillary; Creatinine; Ergocalciferols; Female; Humans; Hypercalcemia; Hypoparathyroidism; Parathyroid Neoplasms; Pharmaceutical Vehicles; Postoperative Complications; Stimulation, Chemical; Thyroid Neoplasms; Thyroidectomy

Porcine calcitonin was used to treat three patients with hypercalcaemia due to vitamin D intoxication. In two patients a rapid and sustained fall to normal in serum calcium occurred within three days, in the third patient normocalcaemia was achieved in seven days. In view of its rapid and sustained effect calcitonin may be of value in the urgent treatment of hypercalcaemia due to vitamin D intoxication.

Topics: Aged; Animals; Back Pain; Calcitonin; Ergocalciferols; Female; Humans; Hypercalcemia; Hypoparathyroidism; Middle Aged; Poisoning; Swine; Vitamin D

Topics: Adolescent; Dihydrotachysterol; Ergocalciferols; Female; Humans; Hypocalcemia; Hypoparathyroidism; Hypophosphatemia, Familial; Infant; Male; Protein-Losing Enteropathies; Pseudohypoparathyroidism

Topics: Blood Urea Nitrogen; Brain; Calcium; Electroencephalography; Ergocalciferols; Female; Humans; Hypercalcemia; Hypocalcemia; Hypoparathyroidism; Middle Aged; Phosphorus; Seizures; Thyroidectomy

Topics: Adult; Aged; Ergocalciferols; Humans; Hypercalcemia; Hyperparathyroidism; Hypocalcemia; Hypoparathyroidism; Ischemia; Middle Aged; Muscular Atrophy; Myelin Sheath; Neoplasms; Peripheral Nerves; Sensation; Vibration

Topics: Cholecalciferol; Ergocalciferols; Humans; Hypercalcemia; Hypoparathyroidism; Vitamin D

Topics: Calcium; Ergocalciferols; Female; Fetal Diseases; Fetus; Genes; Humans; Hyperparathyroidism; Hypocalcemia; Hypoparathyroidism; Infant, Newborn; Parathyroid Hormone; Pregnancy; Pregnancy Complications

Topics: Acupuncture Therapy; Adult; Calcium; Dihydrotachysterol; Ergocalciferols; Female; Humans; Hypocalcemia; Hypoparathyroidism; Middle Aged; Tetany

Topics: Calcium; Ergocalciferols; Female; Humans; Hypoparathyroidism; Infant; Infant, Newborn; Magnesium; Parathyroid Hormone; Pedigree; Seizures; Time Factors

Topics: Calcium; Cholecalciferol; Ergocalciferols; Humans; Hypoparathyroidism; Osteomalacia; Rickets; Vitamin D

Topics: Acute Disease; Aged; Basilar Artery; Calcium; Cerebrovascular Disorders; Diagnosis, Differential; Ergocalciferols; Female; Humans; Hypercalcemia; Hypoparathyroidism; Neurologic Manifestations

Topics: Calcium; Ergocalciferols; Humans; Hypoparathyroidism

Topics: Adult; Calcium; Ergocalciferols; Female; Humans; Hypercalcemia; Hypocalcemia; Hypoparathyroidism; Lactates; Male; Middle Aged

Topics: Adenocarcinoma; Adult; Calcium; Electrocardiography; Electroencephalography; Epilepsy; Ergocalciferols; Humans; Hypocalcemia; Hypoparathyroidism; Lung Neoplasms; Male

Topics: Adult; Aged; Calcium; Dihydrotachysterol; Ergocalciferols; Female; Humans; Hypoparathyroidism; Male; Middle Aged; Parathyroid Glands; Postoperative Complications; Thyroidectomy

Topics: Adolescent; Adrenal Insufficiency; Adult; Child; Child, Preschool; Dihydrotachysterol; Ergocalciferols; Female; Humans; Hypercalcemia; Hypoparathyroidism; Male; Rickets; Vitamin D

Topics: Calcium; Cataract; Ergocalciferols; Female; Humans; Hypoparathyroidism; Intestinal Mucosa; Malabsorption Syndromes; Middle Aged; Phosphorus

Topics: Calcium; Child; Dihydrotachysterol; Electrocardiography; Electromyography; Ergocalciferols; Female; Humans; Hypoparathyroidism; Infant; Infant, Newborn; Infant, Newborn, Diseases; Male; Seizures; Tetany

Topics: Ergocalciferols; Gluconates; Humans; Hypoparathyroidism; Lactates; Thyroid Hormones; Thyroidectomy

Topics: Drug Therapy; Ergocalciferols; Gastrectomy; Humans; Hypocalcemia; Hypoparathyroidism; Postgastrectomy Syndromes

Topics: Adolescent; Bone Neoplasms; Calcinosis; Calcium; Calcium Isotopes; Ergocalciferols; Feces; Geriatrics; Humans; Hyperparathyroidism; Hypoparathyroidism; Inositol; Intestinal Absorption; Malabsorption Syndromes; Neoplasm Metastasis; Neoplasms; Osteitis Deformans; Osteomalacia; Pancreatitis; Sarcoidosis; Tetany; Urine

Topics: Adult; Deafness; Dihydrotachysterol; Ergocalciferols; Female; Humans; Hypoparathyroidism; Ophthalmoplegia; Parathyroid Hormone

Topics: Calcium; Calcium, Dietary; Diet; Diet Therapy; Ergocalciferols; Humans; Hypoparathyroidism; Pseudohypoparathyroidism

Topics: Bone and Bones; Calcium; Calcium Isotopes; Calcium Metabolism Disorders; Dihydrotachysterol; Ergocalciferols; Humans; Hyperparathyroidism; Hypoparathyroidism; Intestine, Small; Intestines; Kidney; Male; Radiometry

Topics: Biomedical Research; Blood; Calcium; Creatine; Creatinine; Ergocalciferols; Female; Humans; Hypoparathyroidism; Infant; Infant, Newborn; Maternal-Fetal Exchange; Menstruation; Phosphates; Pregnancy; Pregnancy Complications; Tetany; Thyroidectomy; Urine

Topics: Dihydrotachysterol; Drug Therapy; Ergocalciferols; Humans; Hyperparathyroidism; Hypocalcemia; Hypoparathyroidism; Tetany

Topics: Calcium; Calcium, Dietary; Child; Diagnosis; Dihydrotachysterol; Drug Therapy; Ergocalciferols; Humans; Hypoparathyroidism

Topics: Absorption; Calcium, Dietary; Digestion; Dihydrotachysterol; Ergocalciferols; Gastric Juice; Hyperparathyroidism; Hypoparathyroidism; Intestine, Small; Intestines; Rats; Research

Topics: Alkaline Phosphatase; Calcium; Calcium Metabolism Disorders; Calcium, Dietary; Ergocalciferols; Hypoparathyroidism; Inulin; Kidney Tubules; Osteitis Fibrosa Cystica; Parathyroid Hormone; Pharmacology; Phosphorus; Pseudohypoparathyroidism; Rats; Urine

Topics: Ergocalciferols; Humans; Hypocalcemia; Hypoparathyroidism; Iatrogenic Disease; Postoperative Complications; Psoriasis; Tetany; Thyroidectomy

Topics: Calcium Metabolism Disorders; Cataract; Dihydrotachysterol; Ergocalciferols; Humans; Hypoparathyroidism; Lens, Crystalline; Tetany

Topics: Administration, Intravenous; Disease; Ergocalciferols; Humans; Hypoparathyroidism; Parathyroid Diseases; Parathyroid Glands; Vitamin D; Vitamins

Topics: Bone and Bones; Ergocalciferols; Humans; Hypoparathyroidism; Parathyroid Glands; Parathyroid Hormone; Vitamin D; Vitamins

Topics: Bone and Bones; Ergocalciferols; Hormones; Humans; Hypoparathyroidism; Parathyroid Glands; Vitamin D; Vitamins

Topics: Calcium; Ergocalciferols; Hypoparathyroidism; Paratyphoid Fever