A novel pathogenic mutation of the CYP27B1 gene in a patient with vitamin D-dependent rickets type 1: a case report

BMC Res Notes. 2014 Nov 5:7:783. doi: 10.1186/1756-0500-7-783.

Abstract

Background: Rickets can occur due to Vitamin D deficiency or defects in its metabolism. Three rare genetic types of rickets with different alterations of genes have been reported, including: Vitamin D dependent rickets type 1, Vitamin D dependent rickets type 2 or also known as Vitamin D resistant rickets and 25 hydroxylase deficiency rickets. Vitamin D dependent rickets type 1 is inherited in an autosomal recessive pattern, and is caused by mutations in the CYP27B1 gene encoding the 1α-hydroxylase enzyme. We report here a new mutation in CYP27B1, which lead to Vitamin D dependent rickets type 1.

Case presentation: We report on a 13-month-old Arabic Saudi girl with Vitamin D dependent rickets type 1 presented with multiple fractures and classic features of rickets. A whole exome sequencing identified a novel pathogenic missense mutation (CYP27B1:Homozygous c.1510C > T(p.Q504X)) which results in a protein truncating alteration. Both parents are heterozygous carriers of the mutation. Based on data search in Human Gene Mutation Database, 63 CYP27B1 alterations were reported: only 28.6% are protein truncating (5 nonsense, 13 frameshift insertions/deletions, 0 gross deletions), while 61.9% are non-truncating (38 missense, 1 small in-frame insertions/deletion), and 9.5% are possible protein-truncating (5 splice, 1 regulatory).

Conclusion: The deleterious effect of this alteration, which was the only mutation detected in the CYP27B1 common gene of Vitamin D dependent rickets type 1 in the proband, and its autosomal recessive inheritance fashion, both support a pathogenic nature of this mutation as the cause of Vitamin D dependent rickets type 1.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • 25-Hydroxyvitamin D3 1-alpha-Hydroxylase / genetics*
  • Arabs / genetics
  • Calcium / therapeutic use
  • DNA Mutational Analysis
  • Databases, Genetic
  • Dietary Supplements
  • Familial Hypophosphatemic Rickets / diagnosis
  • Familial Hypophosphatemic Rickets / drug therapy
  • Familial Hypophosphatemic Rickets / enzymology
  • Familial Hypophosphatemic Rickets / ethnology
  • Familial Hypophosphatemic Rickets / genetics*
  • Female
  • Genetic Predisposition to Disease
  • Heredity
  • Homozygote
  • Humans
  • Infant
  • Mutation, Missense*
  • Pedigree
  • Phenotype
  • Saudi Arabia
  • Vitamin D / therapeutic use

Substances

  • Vitamin D
  • 25-Hydroxyvitamin D3 1-alpha-Hydroxylase
  • Calcium

Supplementary concepts

  • Vitamin D Hydroxylation-Deficient Rickets, Type 1A