Multigene Sequencing Analysis of Children Born Small for Gestational Age With Isolated Short Stature

J Clin Endocrinol Metab. 2019 Jun 1;104(6):2023-2030. doi: 10.1210/jc.2018-01971.

Abstract

Context: Patients born small for gestational age (SGA) who present with persistent short stature could have an underlying genetic etiology that will account for prenatal and postnatal growth impairment. We applied a unique massive parallel sequencing approach in cohort of patients with exclusively nonsyndromic SGA to simultaneously interrogate for clinically substantial genetic variants.

Objective: To perform a genetic investigation of children with isolated short stature born SGA.

Design: Screening by exome (n = 16) or targeted gene panel (n = 39) sequencing.

Setting: Tertiary referral center for growth disorders.

Patients and methods: We selected 55 patients born SGA with persistent short stature without an identified cause of short stature.

Main outcome measures: Frequency of pathogenic findings.

Results: We identified heterozygous pathogenic or likely pathogenic genetic variants in 8 of 55 patients, all in genes already associated with growth disorders. Four of the genes are associated with growth plate development, IHH (n = 2), NPR2 (n = 2), SHOX (n = 1), and ACAN (n = 1), and two are involved in the RAS/MAPK pathway, PTPN11 (n = 1) and NF1 (n = 1). None of these patients had clinical findings that allowed for a clinical diagnosis. Seven patients were SGA only for length and one was SGA for both length and weight.

Conclusion: These genomic approaches identified pathogenic or likely pathogenic genetic variants in 8 of 55 patients (15%). Six of the eight patients carried variants in genes associated with growth plate development, indicating that mild forms of skeletal dysplasia could be a cause of growth disorders in this group of patients.

Publication types

  • Evaluation Study
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Body Height / genetics*
  • Body Weight / genetics
  • Child
  • Child, Preschool
  • Exome Sequencing
  • Female
  • Genetic Markers / genetics
  • Growth Disorders / diagnosis*
  • Growth Disorders / genetics
  • High-Throughput Nucleotide Sequencing*
  • Humans
  • Infant, Newborn
  • Infant, Small for Gestational Age / growth & development*
  • Male

Substances

  • Genetic Markers