Hypophosphatemic Rickets with R179W Mutation in FGFR23 Gene - A Rare But Treatable Cause of Refractory Rickets

Indian J Pediatr. 2021 Jan;88(1):61-63. doi: 10.1007/s12098-020-03335-7. Epub 2020 May 16.

Abstract

Hypophosphatemic rickets is one of the major causes of refractory rickets exhibiting genetic heterogeneity. Most cases are X-linked due to PHEX gene mutations. However recently, autosomal dominant (AD) forms have been described, due to mutations in FGF23. The authors present a 13-year-old girl who had hypophosphatemic rickets due to R179W mutation in FGF23 gene, being the first case in India with this mutation. She presented with bone pains, short stature and osteopenic bones, symptoms appearing after onset of menarche. This presentation is different from that seen in younger children with rickets. Burosumab, an anti-FGF23 antibody is an effective novel therapy for FGF23-related rickets but it is not available in India. High doses of calcitriol and phosphate were required to alleviate the symptoms and signs. The authors aim to alert pediatricians to keep in mind this treatable disorder to prevent diagnostic delays and improve treatment outcome.

Keywords: Calcitriol; FGF23 related; Hypophosphatemic rickets; Phosphate; R179W.

Publication types

  • Case Reports

MeSH terms

  • Adolescent
  • DNA Mutational Analysis
  • Familial Hypophosphatemic Rickets* / diagnosis
  • Familial Hypophosphatemic Rickets* / drug therapy
  • Familial Hypophosphatemic Rickets* / genetics
  • Female
  • Fibroblast Growth Factor-23
  • Fibroblast Growth Factors / genetics*
  • Humans
  • India
  • Mutation
  • Phosphates
  • Rickets, Hypophosphatemic* / diagnosis
  • Rickets, Hypophosphatemic* / drug therapy
  • Rickets, Hypophosphatemic* / genetics

Substances

  • FGF23 protein, human
  • Phosphates
  • Fibroblast Growth Factors
  • Fibroblast Growth Factor-23