Diaphanospondylodysostosis and ischiospinal dysostosis, evidence for one disorder with variable expression in a patient who has survived to age 9 years

Am J Med Genet A. 2017 Oct;173(10):2808-2813. doi: 10.1002/ajmg.a.38395. Epub 2017 Aug 17.

Abstract

Diaphanospondylodysostosis (DSD) and ischiospinal dysostosis (ISD) are both rare skeletal dysplasias consisting of abnormal axial skeletal development but normal appendicular skeletal development. Both disorders recently have been found to result from mutations in the BMPER gene. We report a patient with one deletion and one mutation of the BMPER gene who has features most consistent with DSD but who has survived to age 9 years. Survival suggests that DSD and ISD reflect a spectrum of severity of one disease process.

Keywords: BMPER; DSD; ISD; diaphanospondylodysostosis; ischiospinal dysostosis; ischiovertebral.

Publication types

  • Case Reports

MeSH terms

  • Carrier Proteins / genetics*
  • Child
  • Craniofacial Abnormalities / genetics*
  • Craniofacial Abnormalities / pathology
  • Dysostoses / genetics*
  • Dysostoses / pathology
  • Humans
  • Ischium / pathology*
  • Male
  • Mutation*
  • Prognosis
  • Ribs / abnormalities*
  • Ribs / pathology
  • Spine / abnormalities*
  • Spine / pathology

Substances

  • BMPER protein, human
  • Carrier Proteins

Supplementary concepts

  • Diaphanospondylodysostosis