Hypophosphatemic rickets. Clinical cases

نویسندگان

چکیده

Hypophosphatemic rickets (HR) is a genetically heterogeneous disease that accompanied by hypophosphatemia, skeletal (rickets-like) disorders, growth retardation in children and adults damage of other organs systems. Its incidence 3.9 cases per 100.000 live births, the prevalence ranges from 1.7 100,000 to 4.8 people (children adults).
 The study was aimed on investigation diagnostic features treatment strategy different HR subtypes evaluation effectiveness therapy. Three patients with two families have been described. Laboratory instrumental investigations were used confirm HR. Genetic diagnosis done using next-generation target sequencing (tNGS) (skeletal dysplasia panel).
 General clinical disorders (e.g. severe deformity lower extremities early childhood) observed All had hypophosphatemia normal/slightly elevated or decreased PTH levels. underwent repeated surgical (without appropriate laboratory genetic testing correction phosphorus-calcium metabolism), which turned out be ineffective led disability. In one family, confirmed presence heterozygous pathogenic PHEX variant, — homozygous SLC34A3 variant. Normophosphatemia achieved girl after starting therapy phosphorus supplements. calcitriol, cholecalciferol supplements little effect (P levels increased but normalization not achieved), thus only burosumab proved rational for such patients. Prior any corrective surgery, thorough examination should performed compensate metabolism. Timely initiation pathogenetic conservative can improve bone structure final growth, as well reduce need surgery.

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ژورنال

عنوان ژورنال: Klìnì?na endokrinologìâ ta endokrinna hìrurgìâ

سال: 2022

ISSN: ['2519-2582', '1818-1384']

DOI: https://doi.org/10.30978/cees-2022-1-89