Axial skeleton anterior-posterior patterning is regulated through feedback regulation between Meis transcription factors and retinoic acid

Development. 2021 Jan 4;148(1):dev193813. doi: 10.1242/dev.193813.

Abstract

Vertebrate axial skeletal patterning is controlled by co-linear expression of Hox genes and axial level-dependent activity of HOX protein combinations. MEIS transcription factors act as co-factors of HOX proteins and profusely bind to Hox complex DNA; however, their roles in mammalian axial patterning remain unknown. Retinoic acid (RA) is known to regulate axial skeletal element identity through the transcriptional activity of its receptors; however, whether this role is related to MEIS/HOX activity remains unknown. Here, we study the role of Meis in axial skeleton formation and its relationship to the RA pathway in mice. Meis elimination in the paraxial mesoderm produces anterior homeotic transformations and rib mis-patterning associated to alterations of the hypaxial myotome. Although Raldh2 and Meis positively regulate each other, Raldh2 elimination largely recapitulates the defects associated with Meis deficiency, and Meis overexpression rescues the axial skeletal defects in Raldh2 mutants. We propose a Meis-RA-positive feedback loop, the output of which is Meis levels, that is essential to establish anterior-posterior identities and patterning of the vertebrate axial skeleton.

Keywords: Embryo patterning; Homeodomain; Homeotic transformation; Myogenesis; Skeletal patterning; Vertebral patterning.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Aldehyde Oxidoreductases / deficiency
  • Aldehyde Oxidoreductases / metabolism
  • Alleles
  • Animals
  • Body Patterning* / genetics
  • Bone and Bones / embryology*
  • Bone and Bones / metabolism*
  • Embryo, Mammalian / metabolism
  • Embryonic Development / genetics
  • Feedback, Physiological*
  • Fetus / metabolism
  • Gene Expression Regulation, Developmental*
  • Homeodomain Proteins / genetics
  • Homeodomain Proteins / metabolism*
  • Integrases / metabolism
  • Mice
  • Models, Biological
  • Mutation / genetics
  • Myeloid Ecotropic Viral Integration Site 1 Protein / genetics
  • Myeloid Ecotropic Viral Integration Site 1 Protein / metabolism*
  • RNA, Messenger / genetics
  • RNA, Messenger / metabolism
  • Signal Transduction
  • Tretinoin / metabolism*

Substances

  • Homeodomain Proteins
  • Meis1 protein, mouse
  • Mrg1 protein, mouse
  • Myeloid Ecotropic Viral Integration Site 1 Protein
  • RNA, Messenger
  • Tretinoin
  • Aldehyde Oxidoreductases
  • RALDH2 protein, mouse
  • Cre recombinase
  • Integrases