A new case of osteogenesis imperfecta type VIII and retinal detachment

Am J Med Genet A. 2021 Jan;185(1):238-241. doi: 10.1002/ajmg.a.61934. Epub 2020 Oct 24.

Abstract

Osteogenesis imperfecta (OI) type VIII (OMIM: 610915) is a rare autosomal recessive disorder characterized by white sclerae, severe growth deficiency, and bone fragility. This condition results from pathogenic variants of P3H1, a gene that codes for P3H1, an important protein involved in the prolyl-3-hydroxylation complex required for collagen type I folding. Here, we described a woman with OI type VIII due to a homozygous mutation of c.1914+1G>C (NM_001243246.1) in P3H1 and retinal detachment. We compared our case to five severe OI and retinal detachment cases reported in the literature. The only case previously reported with a molecular diagnosis had a similar mutation in P3H1 c.1914+1G>A and a giant retinal detachment. We suggest that individuals with OI type VIII should be submitted to careful fundoscopic examination.

Keywords: P3H1; osteogenesis imperfecta; retinal detachment.

Publication types

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

MeSH terms

  • Adolescent
  • Adult
  • Child
  • Collagen Type I / genetics
  • Female
  • Fibroblasts / metabolism
  • Genetic Predisposition to Disease*
  • Humans
  • Male
  • Membrane Glycoproteins / genetics*
  • Middle Aged
  • Mutation
  • Osteogenesis Imperfecta / complications
  • Osteogenesis Imperfecta / genetics*
  • Osteogenesis Imperfecta / pathology
  • Prolyl Hydroxylases / genetics*
  • Proteoglycans / genetics*
  • Retinal Detachment / complications
  • Retinal Detachment / genetics*
  • Retinal Detachment / pathology
  • Sclera / pathology
  • Young Adult

Substances

  • Collagen Type I
  • Membrane Glycoproteins
  • Proteoglycans
  • Prolyl Hydroxylases
  • P3H1 protein, human

Supplementary concepts

  • Osteogenesis imperfecta, type VIII