Literature DB >> 3153320

Autosomal dominant hypophosphataemia with elevated serum 1,25 dihydroxyvitamin D and hypercalciuria.

W C Proesmans1, G Fabry, G J Marchal, P L Gillis, R Bouillon.   

Abstract

A 14-year-old boy presented with the clinical and radiological features of rickets. Serum inorganic phosphate levels were constantly low, whereas serum calcium and parathyroid hormone levels were within the normal range. Laboratory investigation did not show any evidence for vitamin-D deficiency, chronic renal insufficiency, Fanconi syndrome, tubular acidosis, hepatic disease or intestinal malabsorption. A family study comprising 34 members over four generations revealed 10 other individuals to be affected and the mode of inheritance to be autosomal dominant. In addition to hypophosphataemia and normocalcaemia, the disease is characterized by elevated serum 1,25 dihydroxyvitamin D levels and hypercalciuria. This hereditary syndrome of renal hypophosphataemia differs from the common familial X-linked hypophosphataemia and the recently described autosomal recessive hypophosphataemic rickets with hypercalciuria by its dominant mode of inheritance; it differs from hypophosphataemic non-rachitic bone disease by the elevated serum 1,25 dihydroxyvitamin D levels and hypercalciuria.

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Year:  1987        PMID: 3153320     DOI: 10.1007/bf00849257

Source DB:  PubMed          Journal:  Pediatr Nephrol        ISSN: 0931-041X            Impact factor:   3.714


  11 in total

1.  Measurement of 25-hydroxyvitamin D3 in serum.

Authors:  R Bouillon; P V Kerkhove; P De Moor
Journal:  Clin Chem       Date:  1976-03       Impact factor: 8.327

2.  A genetic study of familial hypophosphatemia and vitamin D resistant rickets with a review of the literature.

Authors:  R W WINTERS; J B GRAHAM; T F WILLIAMS; V W McFALLS; C H BURNETT
Journal:  Medicine (Baltimore)       Date:  1958-05       Impact factor: 1.889

3.  The measurement of the vitamin D-binding protein in human serum.

Authors:  R Bouillon; H van Baelen; P de Moor
Journal:  J Clin Endocrinol Metab       Date:  1977-08       Impact factor: 5.958

4.  Serum 1,25-dihydroxyvitamin D levels in normal subjects and in patients with hereditary rickets or bone disease.

Authors:  C R Scriver; T M Reade; H F DeLuca; A J Hamstra
Journal:  N Engl J Med       Date:  1978-11-02       Impact factor: 91.245

5.  Direct effects of vitamin D and its metabolites on skeletal tissue.

Authors:  L G Raisz
Journal:  Clin Endocrinol Metab       Date:  1980-03

6.  Hypophosphatemic nonrachitic bone disease: an entity distinct from X-linked hypophosphatemia in the renal defect, bone involvement, and inheritance.

Authors:  C R Scriver; W MacDonald; T Reade; R H Glorieux; B Nogrady
Journal:  Am J Med Genet       Date:  1977

7.  Hereditary hypophosphatemic rickets with hypercalciuria.

Authors:  M Tieder; D Modai; R Samuel; R Arie; A Halabe; I Bab; D Gabizon; U A Liberman
Journal:  N Engl J Med       Date:  1985-03-07       Impact factor: 91.245

8.  Intestinal Ca and phosphate transport: differential responses to vitamin D3 metabolites.

Authors:  M W Walling
Journal:  Am J Physiol       Date:  1977-12

9.  Long-term influence of calcitriol (1,25-dihydroxyvitamin D) and supplemental phosphate in X-linked hypophosphatemic rickets.

Authors:  R W Chesney; R B Mazess; P Rose; A J Hamstra; H F DeLuca; A L Breed
Journal:  Pediatrics       Date:  1983-04       Impact factor: 7.124

10.  A radioimmunoassay for 1,25-dihydroxycholecalciferol.

Authors:  R Bouillon; P De Moor; E G Baggiolini; M R Uskokovic
Journal:  Clin Chem       Date:  1980-04       Impact factor: 8.327

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  4 in total

Review 1.  Hypophosphatemic rickets.

Authors:  L A DiMeglio; M J Econs
Journal:  Rev Endocr Metab Disord       Date:  2001-04       Impact factor: 6.514

2.  Targeted inactivation of Npt2 in mice leads to severe renal phosphate wasting, hypercalciuria, and skeletal abnormalities.

Authors:  L Beck; A C Karaplis; N Amizuka; A S Hewson; H Ozawa; H S Tenenhouse
Journal:  Proc Natl Acad Sci U S A       Date:  1998-04-28       Impact factor: 11.205

3.  A girl with rickets and nephrocalcinosis.

Authors:  Nathalie Godefroid; Willem Proesmans
Journal:  Pediatr Nephrol       Date:  2003-04-29       Impact factor: 3.714

4.  Family investigations in idiopathic hypercalciuria.

Authors:  F Harangi; K Méhes
Journal:  Eur J Pediatr       Date:  1993-01       Impact factor: 3.183

  4 in total

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