Trabecular bone microarchitecture analysis, a way for an early detection of genetic dwarfism? Case study of a dwarf mother's offspring

Int J Paleopathol. 2018 Mar:20:65-71. doi: 10.1016/j.ijpp.2017.12.002. Epub 2017 Dec 14.

Abstract

A 66 year-old woman with a disproportionate dwarfism and who bore seven children was discovered at the Middenbeemster archaeological site (The Netherlands). Three are perinates and show no macroscopic or radiological evidence for a FGFR3 mutation causing hypo-or achondroplasia. This mutation induces dysfunction of the growth cartilage, leading to abnormalities in the development of trabecular bone. Because the mutation is autosomal dominant, these perinates have a 50% risk of having been affected. This study determines whether trabecular bone microarchitecture (TBMA) analysis is useful for detecting genetic dwarfism. Proximal metaphyses of humeri were μCT-scanned with a resolution of 7-12 μm. Three volumes of interest were segmented from each bone with TIVMI© software. The TBMA was quantified in BoneJ© using six parameters on which a multivariate analysis was then performed. Two of the Middenbeemster perinates show a quantitatively different TBMA organization. These results and the family's medical history suggest a diagnosis of genetic dwarfism for this two perinates. This study provides evidence to support the efficacy of μCT for diagnosing early-stage bone disease.

Keywords: Genetic dwarfism; Human growth; Micro-computed tomography; Trabecular bone microarchitecture.

Publication types

  • Historical Article
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Achondroplasia / diagnostic imaging
  • Achondroplasia / genetics
  • Achondroplasia / history
  • Achondroplasia / pathology
  • Aged
  • Cancellous Bone / abnormalities
  • Cancellous Bone / diagnostic imaging
  • Dwarfism* / diagnostic imaging
  • Dwarfism* / genetics
  • Dwarfism* / history
  • Dwarfism* / pathology
  • Female
  • History, 19th Century
  • Humans
  • Humerus / abnormalities
  • Humerus / diagnostic imaging
  • Humerus / pathology
  • Infant
  • Mothers
  • Mutation
  • Netherlands
  • Paleopathology
  • Receptor, Fibroblast Growth Factor, Type 3* / genetics
  • X-Ray Microtomography

Substances

  • FGFR3 protein, human
  • Receptor, Fibroblast Growth Factor, Type 3