Molecular screening of PROKR2 gene in girls with idiopathic central precocious puberty

Ital J Pediatr. 2021 Jan 7;47(1):5. doi: 10.1186/s13052-020-00951-z.

Abstract

Background: Prokineticin receptor 2 (PROKR2) loss of function mutations have been described as cause of hypogonadotropic hypogonadism. In 2017, a first case of central precocious puberty (CPP) caused by PROKR2 heterozygous gain of function mutation was described in a 3.5 years-old girl. No other cases have been reported yet. This study performs a molecular screening in girls with early onset CPP (breast budding before 6 years of age) to identify possible alterations in PROKR2.

Methods: We analysed DNA of 31 girls with idiopathic CPP diagnosed via basal LH levels > 0.3 IU/L or peak-LH > 5 IU/L after stimulation, without any MKRN3 mutations. The Fisher exact test was used to compare polymorphism allele frequency to corresponding ones in genome aggregation database (gnomAD).

Results: No rare variants were identified. Five polymorphisms were found (rs6076809, rs8116897, rS3746684, rs3746682, rs3746683). All except one (i.e. rs3746682) had a minor allele frequency (MAF) similar to that reported in literature. rs3746682 presented a MAF higher than that described in the gnomAD (0.84 in our cohort vs 0.25 from gnomAD).

Conclusions: As for other G protein-coupled receptors (i.e. GPR54), mutations in PROKR2 do not seem to be a frequent cause of CPP in girls.

Keywords: Early central precocious puberty; Genetic screening; PROKR2.

Publication types

  • Multicenter Study
  • Observational Study

MeSH terms

  • Child, Preschool
  • Cohort Studies
  • Female
  • Genetic Testing
  • Humans
  • Infant
  • Italy
  • Mutation / genetics*
  • Polymorphism, Genetic / genetics*
  • Puberty, Precocious / diagnosis
  • Puberty, Precocious / genetics*
  • Receptors, G-Protein-Coupled / genetics*
  • Receptors, Peptide / genetics*

Substances

  • PROKR2 protein, human
  • Receptors, G-Protein-Coupled
  • Receptors, Peptide