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Undetectable serum calcidiol: not everything that glitters is gold.

Gallegos-Bayas G, Pascual-Pareja JF, Sanchez-Niño MD, Manzarbeitia F, Ortiz A - Clin Kidney J (2012)

Bottom Line: There is an increased awareness of the adverse consequences of nutritional vitamin D deficiency.Upon recovery, serum 1,25(OH)2 vitamin D remained in the normal range despite CKD and serum 25(OH) vitamin D 6 ng/mL.This case illustrates the dangers of supplementing vitamin D in patients with low serum 25(OH) vitamin D and increased 1α-hydroxylase activity due to granulomatous disease.

View Article: PubMed Central - PubMed

Affiliation: Department of Family and Community Medicine, Centro de Salud Barrio del Pilar, Area 5, Madrid, Spain.

ABSTRACT
There is an increased awareness of the adverse consequences of nutritional vitamin D deficiency. We report a patient with chronic tophaceous gout, chronic kidney disease (CKD) Stage 3/4 and undetectable serum calcidiol who developed severe hypercalcaemia upon vitamin D supplementation despite serum 25(OH) vitamin D within the normal range. Upon recovery, serum 1,25(OH)2 vitamin D remained in the normal range despite CKD and serum 25(OH) vitamin D 6 ng/mL. Gout tophi biopsies from additional patients showed macrophage expression of 25(OH) vitamin D 1α-hydroxylase. This case illustrates the dangers of supplementing vitamin D in patients with low serum 25(OH) vitamin D and increased 1α-hydroxylase activity due to granulomatous disease.

No MeSH data available.


Related in: MedlinePlus

Macrophage expression of 1α-hydroxylase. (A) Gouty tophi stained with H&E. Granulomatous inflammation is observed surrounding the matrix in which urate crystals, dissolved during the deprocessing procedure, were originally embedded. Original magnification ×100. (B) Representative image of 1α-hydroxylase-expressing histiocytic mononucleated and multinucleated giant cells (arrows) in gouty tophi, as assessed by immunohistochemistry. Original magnification ×100, detail ×400. (C) 24-Hydroxylase-expressing ‘macrophagic’ mononucleated and multinucleated giant cells (arrows) in gouty tophi, as assessed by immunohistochemistry. Original magnification ×100. (D) CD68 staining confirmed the macrophagic nature of cells surrounding the urate core (arrows) in gouty tophi. Original magnification ×100.
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fig2: Macrophage expression of 1α-hydroxylase. (A) Gouty tophi stained with H&E. Granulomatous inflammation is observed surrounding the matrix in which urate crystals, dissolved during the deprocessing procedure, were originally embedded. Original magnification ×100. (B) Representative image of 1α-hydroxylase-expressing histiocytic mononucleated and multinucleated giant cells (arrows) in gouty tophi, as assessed by immunohistochemistry. Original magnification ×100, detail ×400. (C) 24-Hydroxylase-expressing ‘macrophagic’ mononucleated and multinucleated giant cells (arrows) in gouty tophi, as assessed by immunohistochemistry. Original magnification ×100. (D) CD68 staining confirmed the macrophagic nature of cells surrounding the urate core (arrows) in gouty tophi. Original magnification ×100.

Mentions: Based on the presence of a chronic granulomatous disease, increased ACE levels [3], hypercalcaemia despite normal 25(OH) vitamin D levels and, later in the disease, normal 1,25(OH)2 vitamin D levels despite CKD and severe 25(OH) vitamin D deficiency, it was hypothesized that the patient had increased extrarenal 1α-hydroxylase activity. However, there were no prior reports of 1α-hydroxylase expression in tophi. Thus, we performed immunohistochemistry for 1α-hydroxylase in biopsies from three additional tophaceous gout patients, which showed intense 1α-hydroxylase expression by macrophages and multinucleated cells surrounding tophi (Figure 2B). Furthermore, these cells stained for 24-hydroxylase (Figure 2C) and were confirmed to be CD68-positive macrophages (Figure 2D).


Undetectable serum calcidiol: not everything that glitters is gold.

Gallegos-Bayas G, Pascual-Pareja JF, Sanchez-Niño MD, Manzarbeitia F, Ortiz A - Clin Kidney J (2012)

Macrophage expression of 1α-hydroxylase. (A) Gouty tophi stained with H&E. Granulomatous inflammation is observed surrounding the matrix in which urate crystals, dissolved during the deprocessing procedure, were originally embedded. Original magnification ×100. (B) Representative image of 1α-hydroxylase-expressing histiocytic mononucleated and multinucleated giant cells (arrows) in gouty tophi, as assessed by immunohistochemistry. Original magnification ×100, detail ×400. (C) 24-Hydroxylase-expressing ‘macrophagic’ mononucleated and multinucleated giant cells (arrows) in gouty tophi, as assessed by immunohistochemistry. Original magnification ×100. (D) CD68 staining confirmed the macrophagic nature of cells surrounding the urate core (arrows) in gouty tophi. Original magnification ×100.
© Copyright Policy - creative-commons
Related In: Results  -  Collection

License
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getmorefigures.php?uid=PMC4400450&req=5

fig2: Macrophage expression of 1α-hydroxylase. (A) Gouty tophi stained with H&E. Granulomatous inflammation is observed surrounding the matrix in which urate crystals, dissolved during the deprocessing procedure, were originally embedded. Original magnification ×100. (B) Representative image of 1α-hydroxylase-expressing histiocytic mononucleated and multinucleated giant cells (arrows) in gouty tophi, as assessed by immunohistochemistry. Original magnification ×100, detail ×400. (C) 24-Hydroxylase-expressing ‘macrophagic’ mononucleated and multinucleated giant cells (arrows) in gouty tophi, as assessed by immunohistochemistry. Original magnification ×100. (D) CD68 staining confirmed the macrophagic nature of cells surrounding the urate core (arrows) in gouty tophi. Original magnification ×100.
Mentions: Based on the presence of a chronic granulomatous disease, increased ACE levels [3], hypercalcaemia despite normal 25(OH) vitamin D levels and, later in the disease, normal 1,25(OH)2 vitamin D levels despite CKD and severe 25(OH) vitamin D deficiency, it was hypothesized that the patient had increased extrarenal 1α-hydroxylase activity. However, there were no prior reports of 1α-hydroxylase expression in tophi. Thus, we performed immunohistochemistry for 1α-hydroxylase in biopsies from three additional tophaceous gout patients, which showed intense 1α-hydroxylase expression by macrophages and multinucleated cells surrounding tophi (Figure 2B). Furthermore, these cells stained for 24-hydroxylase (Figure 2C) and were confirmed to be CD68-positive macrophages (Figure 2D).

Bottom Line: There is an increased awareness of the adverse consequences of nutritional vitamin D deficiency.Upon recovery, serum 1,25(OH)2 vitamin D remained in the normal range despite CKD and serum 25(OH) vitamin D 6 ng/mL.This case illustrates the dangers of supplementing vitamin D in patients with low serum 25(OH) vitamin D and increased 1α-hydroxylase activity due to granulomatous disease.

View Article: PubMed Central - PubMed

Affiliation: Department of Family and Community Medicine, Centro de Salud Barrio del Pilar, Area 5, Madrid, Spain.

ABSTRACT
There is an increased awareness of the adverse consequences of nutritional vitamin D deficiency. We report a patient with chronic tophaceous gout, chronic kidney disease (CKD) Stage 3/4 and undetectable serum calcidiol who developed severe hypercalcaemia upon vitamin D supplementation despite serum 25(OH) vitamin D within the normal range. Upon recovery, serum 1,25(OH)2 vitamin D remained in the normal range despite CKD and serum 25(OH) vitamin D 6 ng/mL. Gout tophi biopsies from additional patients showed macrophage expression of 25(OH) vitamin D 1α-hydroxylase. This case illustrates the dangers of supplementing vitamin D in patients with low serum 25(OH) vitamin D and increased 1α-hydroxylase activity due to granulomatous disease.

No MeSH data available.


Related in: MedlinePlus