Treatment of osteomalacia associated with primary biliary cirrhosis with parenteral vitamin D2 or oral 25-hydroxyvitamin D3.

Compston, J.E.; Horton, L. W. L.; Thompson, R P

doi:10.1136/gut.20.2.133

Cited by 32 publications

(6 citation statements)

References 16 publications

(16 reference statements)

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“…Until fully studied in CF, this approach will require close monitoring to ensure hypervitaminosis D or hypercalcemia does not result. Alternatively, intramuscular ergocalciferol may be considered, although a previous study in CF failed to note a significant rise in serum 25-OHD levels 2 weeks after a single injection of 500,000 IU (29,30).…”

Section: Discussionmentioning

confidence: 99%

Failure of High-Dose Ergocalciferol to Correct Vitamin D Deficiency in Adults with Cystic Fibrosis

Boyle

Noschese

Watts

et al. 2005

Am J Respir Crit Care Med

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Survival has steadily improved in cystic fibrosis (CF) over the last few decades, with the median survival age now older than 33 years, and over 40% of all individuals with CF older than 18 years (1). With this increase in survival, there has also been growing recognition of health issues unique to the adult population with CF (2). One of these issues is bone health, because studies using dual-energy x-ray absorptiometry measurements of bone density have determined that, despite their young age, approximately 20 to 25% of adults with CF have osteoporosis and another 40% have osteopenia (3-5). The etiology of this bone disease is multifactorial, with contributors including fat malabsorption (resulting in vitamin D malabsorption and abnormal calcium metabolism) (6-8), glucocorticoid therapy (9, 10), poor nutritional status (11), inadequate gonadal hormones (10, 12), and elevated circulating cytokines (13). Recognition of this increased risk of osteoporosis in CF has led to greater efforts to optimize vitamin D status and calcium absorption (4,14). The overall prevalence of vitamin D deficiency in adults with CF is strikingly high, with as many as 50 to 60% having 25-hydroxyvitamin D (25-OHD) levels below 20 ng/ml (5, 15). Data demonstrating that serum 25-OHD levels need to exceed 30 ng/ml to prevent a rise in serum parathyroid hormone (16) suggest that the prevalence of vitamin D deficiency in CF may be even higher.Because of the growing concern about bone disease in CF, the U.S. Cystic Fibrosis Foundation (CFF) convened a panel of experts to develop consensus treatment guidelines to optimize CF bone health, and these recommendations were recently published (17). The guidelines contained several specific recommendations regarding vitamin D. First, greater attention to maintaining adequate serum 25-OHD levels should occur, with levels being checked annually in the late fall with the goal of maintaining serum 25-OHD between 30 and 60 ng/ml (75-150 nmol/L). Second, all individuals with CF who are older than 1 year should receive 800 IU/day of oral ergocalciferol (vitamin D 2 ). Failure of this supplementation to maintain serum 25-OHD levels above 30 ng/ml should lead to aggressive repletion with high-dose oral ergocalciferol. The guidelines recommended a stepped approach for repletion, with an initial regimen of 50,000 IU/week of oral ergocalciferol for 8 weeks. Failure of this regimen to raise serum 25-OHD to over 30 ng/ml should lead to a second course of oral ergocalciferol with 50,000 IU/twice weekly for 8 weeks. Persistence of serum 25-OHD levels below 30 ng/ml despite these supplemental regimens should result in consideration of alternative modes of therapy, such as calcitriol or phototherapy. This treatment protocol was developed on the basis of data in individuals without CF, but was recognized as a "first step" in addressing the clear need for more aggressive vitamin D therapy in individuals with CF. Conference leaders stated a need for CF-specific data in the future on which to determine optimal dosing rec...

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Section: Discussionmentioning

confidence: 99%

Failure of High-Dose Ergocalciferol to Correct Vitamin D Deficiency in Adults with Cystic Fibrosis

Boyle

Noschese

Watts

et al. 2005

Am J Respir Crit Care Med

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“…Elevation of serum PTH and osteocalcin levels in some patients suggests continuing bone remodeling. Copyright 1999 by the American Association for the Study of Liver Diseases B one disease is well recognized in patients with chronic liver disease [1][2][3][4][5][6][7][8][9][10][11][12][13][14] and is usually caused by low bone-turnover osteoporosis. 5,8,9,12 Although orthotopic liver transplantation (OLT) may stabilize or reverse this process, there is further significant postoperative bone loss.…”

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confidence: 99%

Evidence of continuing bone recovery at a mean of 7 years after liver transplantation

et al. 1999

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Patients with end-stage liver disease have low bone-turnover osteoporosis, and there is often further bone loss of 20% to 30% after orthotopic liver transplantation (OLT). Bone recovery after OLT has been reported, but data are limited. We undertook studies to determine whether bone recovery continues in the long term. Twenty-eight adult patients alive at least 5 years after OLT were studied (14 men, 14 women). Bone mineral density (BMD), serum parathyroid hormone (PTH), osteocalcin, and vitamin D levels were measured pretransplantation, at 3 months, 12 months, a mean of 46 months, and a mean of 85 months (range, 63 to 117 months) after transplantation. When BMD is expressed as a z score, the results were as follows: ؋0.82 ؎ 0.22 pre-OLT; ؊2.04 ؎ 0.27 at 3 months; ؊1.68 ؎ 0.24 at 12 months; ؊1.23 ؎ 0.24 at a mean of 46 months; and ؊1.0 ؎ 0.26 at a mean of 85 months after OLT. The results at 46 and 85 months were significantly greater than the measurement at 3 months after OLT (P F .05). Furthermore, mean BMD (expressed as a z score) returns to the pre-OLT level at a mean of 85 months. At final follow-up, 9 of 28 patients had elevated PTH levels, and 14 of 27 patients had elevated osteocalcin levels. Five patients had spontaneous fractures in the first 12 months after transplantation, and 5 more patients had fractures by final follow-up. Even at 7 years after OLT, there was a significant increase in BMD (expressed as a z score) compared with 3 months after transplantation. Elevation of serum PTH and osteocalcin levels in some patients suggests continuing bone remodeling.

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“…Serum 25-hydroxyvitamin D levels are often low in patients with chronic cholestatic liver disease, such as primary biliary cirrhosis (1)(2)(3)(4)(5)(6) and biliary atresia (7, 8), and metabolic bone disease is a frequent and severe complication of these disorders. Vitamin D malabsorption associated with intraluminal bile salt deficiency is an important contributing factor to vitamin D deficiency in cholestatic syndromes (9)(10)(11)(12).…”

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confidence: 99%

Iepatic vitamin D 25-hydroxylase: Inhibition by bile duct ligation or bile salts

et al. 1981

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Bone disease and low serum levels of 25-hydroxyvitamin D are prevalent in cholestatic syndromes such as primary biliary cirrhosis and biliary atresia. Defective hydroxylation, along with malabsorption of vitamin D, could be a factor in 25-hydroxyvitamin D depletion. To assess hepatic hydroxylation during experimental cholestasis, we studied vitamin D 25-hydroxylase activity in liver homogenates of rats after 7, 14, and 21 days of bile duct ligation. We have also studied the effects of bile acids on this enzyme in vitro. Hepatic 25-hydroxylation was depressed after 7 days ligation in only 1 of 4 animals, but by 14 days, all animals showed a marked reduction with a mean decrease of 64% in specific activity. Total liver enzyme activity was reduced by 43% at 14 days. In the ligated animals, liver histology showed progressive bile stasis, focal necrosis, bile ductular proliferation, periductular and periportal inflammation, and fibrosis. Addition of bile acids to the in vitro assay in concentrations approximating those found in cholestasis produced marked inhibition of vitamin D 25-hydroxylase activity.

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Treatment of osteomalacia associated with primary biliary cirrhosis with parenteral vitamin D2 or oral 25-hydroxyvitamin D3.

Cited by 32 publications

References 16 publications

Failure of High-Dose Ergocalciferol to Correct Vitamin D Deficiency in Adults with Cystic Fibrosis

Failure of High-Dose Ergocalciferol to Correct Vitamin D Deficiency in Adults with Cystic Fibrosis

Evidence of continuing bone recovery at a mean of 7 years after liver transplantation

Iepatic vitamin D 25-hydroxylase: Inhibition by bile duct ligation or bile salts

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