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Dental lesions in tumoral calcinosis (1991)

Burkes, E. Jeff ; Lyles, Kenneth W. ; Dolan, Edward A. ; [et al.]

Oxford, UK : Blackwell Publishing Ltd

Journal of oral pathology & medicine 20 (1991), S. 0

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ISSN: 1600-0714

Source: Blackwell Publishing Journal Backfiles 1879-2005

Topics: Medicine

Notes: Tumoral calcinosis (TC) is a rare inherited autosomal dominant metabolic disease manifested by elevated serum phosphorus and 1.25 dihydroxyvitamin D levels and periarticular cystic and solid tumourous calcifications. The dental findings in a large family have been critical in determining the genetic transmission of the condition. Radiographically the teeth have short bulbous roots, pulp stones and partial obliteration of the pulp cavity. Histologically, coronal dentin and a variable amount of radicular dentin appears to be deposited regularly. At nonspecific points the developing radicular dentin appears to encounter a mass of calcified material and proceed to grow around it. This mass has a unique histologic pattern with ovoid spaces surrounded by amorphous calcification. At levels of further root development the radicular dentin has an irregular bending tubule arrangement. The dental lesion of TC appears to be different from that of radicular dentin dysplasia in histologic structure and in the method of initiation of the dentin defect. These data suggest that the specific dental lesion is a new phenotypic marker for TC.

Type of Medium: Electronic Resource

URL: http://dx.doi.org/10.1111/j.1600-0714.1991.tb00423.x

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Serum 1,25-dihydroxyvitamin D levels in subjects with X-linked hypophosphatemic rickets and osteomalacia (1982)

Lyles, Kenneth W. ; Clark, Ann G. ; Drezner, Marc K.

Springer

Calcified tissue international 34 (1982), S. 125-130

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ISSN: 1432-0827

Keywords: X-Linked hypophosphatemic rickets ; Osteomalacia ; 1,25-Dihydroxyvitamin D ; Bone

Source: Springer Online Journal Archives 1860-2000

Topics: Biology , Medicine , Physics

Notes: Summary In order to determine whether a defect in vitamin D metabolism might play a role in the pathogenesis of X-linked hypophosphatemic rickets and osteomalacia (XLH), we compared the serum 1,25-dihydroxyvitamin D [1,25(OH)2D] level in 52 normal subjects and 37 patients with XLH. In untreated patients, adults were found to have values similar to age-matched controls, while youths had values similar to growth-rate-matched controls but significantly lower than the levels of age-matched controls who were growing at a normal rate. In contrast, treated XLH patients of all ages had serum levels significantly lower than both controls and untreated XLH patients. Further, the serum levels of 1,25(OH)2D in these treated patients had a significant inverse linear correlation with serum 25-(OH)D concentrations. We propose that subjects with XLH have serum 1,25(OH)2D levels within appropriate age- and growth-rate-matched normal ranges. However, in the presence of hypophosphatemia, we would have anticipated elevated levels of 1,25(OH)2D; viewed in this light the serum 1,25(OH)2D levels are inadequate, suggesting the presence of a relative deficiency of this active vitamin D metabolite.