Impaired response to acute phosphorus (P) deprivation in familial hypophosphatemic rickets (HR)

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چکیده

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Impaired phosphorus conservation and 1,25 dihydroxyvitamin D generation during phosphorus deprivation in familial hypophosphatemic rickets.

The pathogenesis of familial hypophosphatemic rickets (FHR) is incompletely understood. We therefore examined the effects of acute dietary phosphorus deprivation to see whether renal phosphate conservation and increased 1,25 dihydroxyvitamin D [1,25(OH)2D] plasma levels, which normally follow restriction of phosphorus intake, could be induced in patients with FHR. Six healthy male volunteers (a...

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Familial Hypophosphatemic Rickets: Pathophysiology and Medical Management

Chronic hypophosphatemia is one major cause of rickets and osteomalacia in growing children (see Preface of this volume). There are acquired and congenital forms. In most instances, the acquired forms can be controlled by acting on the underlying cause (insufficient phosphate intake, increased renal loss secondary to a mesenchymal tumor, or an altered renal tubular function). The inherited synd...

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Dentin noncollagenous matrix proteins in familial hypophosphatemic rickets.

Familial hypophosphatemic rickets is transmitted in most cases as an X-linked dominant trait and results from the mutation of the PHEX gene predominantly expressed in osteoblast and odontoblast. Patients with rickets have been reported to display important dentin defects. Our purpose was to explore the structure, composition and distribution of noncollagenous proteins (NCPs) of hypophosphatemic...

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Tertiary hyperparathyroidism during high phosphate therapy of familial hypophosphatemic rickets.

We report the development of severe tertiary hyperparathyroidism in three girls treated for familial hypophosphatemic rickets and characterize parathyroid function in vivo and in vitro. All patients had been previously treated with relatively large doses of inorganic phosphorus (125 mm/day) and ergocalciferol or calcitriol for several years and had radiographic evidence of long-standing hyperpa...

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Familial Hypophosphatemic Rickets - A Case Report and Review of Literature

Introduction Familial hypophosphatemic or X-linked hypophosphatemic (XLH) rickets is the most common form of non-nutritional rickets1. The prevalence of XLH rickets yet remain unknown in Bangladesh. It is an Xlinked dominant disorder characterized by renal phosphate wasting with consequent defect of bone mineralization1. Some form of the disease are observed to be transmitted which followed an ...

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ژورنال

عنوان ژورنال: Pediatric Research

سال: 1981

ISSN: 0031-3998,1530-0447

DOI: 10.1203/00006450-198112000-00045