Preimplantation genetic diagnosis for severe albright hereditary osteodystrophy

J Clin Endocrinol Metab. 2008 Mar;93(3):901-4. doi: 10.1210/jc.2007-2040. Epub 2007 Dec 18.

Abstract

Context: Preimplantation genetic diagnosis (PGD) enables the selection of embryos without mutations for implantation and has not been described to our knowledge for mutations in GNAS. Phocomelia in a patient with Albright hereditary osteodystrophy (AHO) has also not been previously described.

Objective: The aim of this study was to identify a GNAS mutation in a patient with a severe form of AHO and pseudohypoparathyroidism type 1a with phocomelia and to perform PGD on embryos derived by in vitro fertilization to deliver an unaffected infant.

Design: A proband and his family are described clinically, the GNAS gene was sequenced to identify a novel mutation in the proband, and PGD was performed on embryos.

Setting: The setting was in a tertiary-care hospital.

Patients: The patients were from a single family in which the proband has a severe form of AHO.

Interventions: Interventions were PGD and in vitro fertilization.

Main outcome measures: The main outcome measures were the clinical phenotypes and GNAS gene sequences of the proband, embryos, and family members.

Results: After PGD, three genotypically normal embryos were transferred back to the mother. Pregnancy ensued, and a healthy male infant was delivered at 36.5 wk gestation. The GNAS genes in the baby were confirmed as wild-type, and the infant is free of any signs of AHO.

Conclusions: We describe herein a proband with AHO and severe skeletal deformities (including phocomelia) related to a novel GNAS mutation and the delivery of a male infant with homozygous normal GNAS genotype after PGD.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Chromogranins
  • Fibrous Dysplasia, Polyostotic / diagnosis
  • Fibrous Dysplasia, Polyostotic / genetics*
  • GTP-Binding Protein alpha Subunits, Gs / genetics*
  • Humans
  • Male
  • Mutation*
  • Preimplantation Diagnosis*

Substances

  • Chromogranins
  • GNAS protein, human
  • GTP-Binding Protein alpha Subunits, Gs