Cell and Tissue Research

, Volume 348, Issue 1, pp 131–140

Trps1 is necessary for normal temporomandibular joint development

  • Ikumi Michikami
  • Toshiya Fukushi
  • Shiho Honma
  • Seisuke Yoshioka
  • Shunji Itoh
  • Yasuteru Muragaki
  • Kojiro Kurisu
  • Takashi Ooshima
  • Satoshi Wakisaka
  • Makoto Abe
Regular Article

DOI: 10.1007/s00441-012-1372-1

Cite this article as:
Michikami, I., Fukushi, T., Honma, S. et al. Cell Tissue Res (2012) 348: 131. doi:10.1007/s00441-012-1372-1

Abstract

Mutation of the human TRPS1 gene leads to trichorhinophalangeal syndrome (TRPS), which is characterized by an abnormal development of various organs including the craniofacial skeleton. Trps1 has recently been shown to be expressed in the jaw joints of zebrafish; however, whether Trps1 is expressed in the mammalian temporomandibular joint (TMJ), or whether it is necessary for TMJ development is unknown. We have analyzed (1) the expression pattern of Trps1 during TMJ development in mice and (2) TMJ development in Trps1 knockout animals. Trps1 is expressed in the maxillo-mandibular junction at embryonic day (E) 11.5. At E15.5, expression is restricted to the developing condylar cartilage and to the surrounding joint disc progenitor cells. In Trps1 knockout mice, the glenoid fossa of the temporal bone forms relatively normally but the condylar process is extremely small and the joint disc and cavities do not develop. The initiation of condyle formation is slightly delayed in the mutants at E14.5; however, at E18.5, the flattened chondrocyte layer is narrowed and most of the condylar chondrocytes exhibit precocious chondrocyte maturation. Expression of Runx2 and its target genes is expanded toward the condylar apex in the mutants. These observations underscore the indispensable role played by Trps1 in normal TMJ development in supporting the differentiation of disc and synoviocyte progenitor cells and in coordinating condylar chondrocyte differentiation.

Keywords

TRPS1TemporomandibularDiscRunx2Mouse

Supplementary material

441_2012_1372_Fig8_ESM.jpg (113 kb)
Supplementary Fig. 1

Normal formation of incudo-malleal and incudo-stapedial junctions in Trps1 knockout mice at E18.5. Hematoxylin-eosin-stained coronal sections of incudo-malleal (a, b) and incudo-stapedial (c, d) junctions in the wild-type (a, c) and mutant (b, d) embryos (eam external acoustic meatus, in cartilage primordium of incus, m cartilage primordium of malleus, mc Meckel’s cartilage, scc superior semicircular canal, st cartilage primordium of stapes). ×50 (JPEG 112 kb)

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Supplementary Fig. 2

In situ hybridization for collagen genes in Trps1 knockout condylar cartilage at E18.5. Serial coronal sections of the wild-type (a, c, e) and mutant (b, d, f) embryos. In situ hybridization for Col1a1 (a, b), Col2a1 (c, d) and Col10a1 (e, f). The dotted line in c-f indicates the margin of the condyle. The solid line in e, f indicates the Col10a1-negative region in the condylar cartilage. ×100 (JPEG 68 kb)

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Supplementary Fig. 3

Alizarin red staining and gene expression analysis in Trps1 knockout condylar cartilage at E18.5. The dotted line in the image indicates the margin of the condyle. Alizarin red staining (a, b) and in situ hybridization (c-h) of the wild-type (a, c, e, g) and mutant (b, d, f, h) embryos. In situ hybridization on coronal sections for Runx2 (c, d), Ihh (e, f) and Ptc1 (g, h) genes (cd condyle, d joint disc, lc lower joint cavity, lpm lateral pterygoid muscle, uc upper joint cavity). ×100 (JPEG 105 kb)

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High resolution image file (TIFF 24659 kb)

Copyright information

© Springer-Verlag 2012

Authors and Affiliations

  • Ikumi Michikami
    • 1
    • 2
  • Toshiya Fukushi
    • 1
    • 5
  • Shiho Honma
    • 1
  • Seisuke Yoshioka
    • 3
  • Shunji Itoh
    • 4
  • Yasuteru Muragaki
    • 4
  • Kojiro Kurisu
    • 5
  • Takashi Ooshima
    • 2
  • Satoshi Wakisaka
    • 1
  • Makoto Abe
    • 1
  1. 1.Department of Oral Anatomy and Developmental BiologyOsaka University Graduate School of DentistryOsakaJapan
  2. 2.Department of Pediatric DentistryOsaka University Graduate School of DentistryOsakaJapan
  3. 3.Department of Restorative DentistryOsaka University Graduate School of DentistryOsakaJapan
  4. 4.First Department of PathologyWakayama Medical University Medical SchoolWakayamaJapan
  5. 5.Department of Judo TherapyYukioka School of Allied Health ProfessionsOsakaJapan