Ift88 limits bone formation in maxillary process through suppressing apoptosis

Momoko Watanabe, Maiko Kawasaki, Katsushige Kawasaki, Atsushi Kitamura, Takahiro Nagai, Yasumitsu Kodama, Fumiya Meguro, Akane Yamada, Paul T. Sharpe, Takeyasu Maeda, Ritsuo Takagi, Atsushi Ohazama*

*Corresponding author for this work

Research output: Contribution to journalArticlepeer-review

9 Citations (Scopus)


Objective: The development of the maxillary bone is under strict molecular control because of its complicated structure. Primary cilia play a critical role in craniofacial development, since defects in primary cilia are known to cause congenital craniofacial dysmorphologies as a wide spectrum of human diseases: the ciliopathies. The primary cilia also are known to regulate bone formation. However, the role of the primary cilia in maxillary bone development is not fully understood. Design: To address this question, we generated mice with a mesenchymal conditional deletion ofIft88 using the Wnt1Cre mice (Ift88 fl/fl ;Wnt1Cre). The gene Ift88 encodes a protein that is required for the function and formation of primary cilia. Results: It has been shown thatIft88 fl/fl ;Wnt1Cre mice exhibit cleft palate. Here, we additionally observed excess bone formation in the Ift88 mutant maxillary process. We also found ectopic apoptosis in the Ift88 mutant maxillary process at an early stage of development. To investigate whether the ectopic apoptosis is related to the Ift88 mouse maxillary phenotypes, we generated Ift88 fl/fl ;Wnt1Cre;p53 −/− mutants to reduce apoptosis. The Ift88 fl/fl ;Wnt1Cre;p53 −/− mice showed no excess bone formation, suggesting that the cells evading apoptosis by the presence of Ift88 in wild-type mice limit bone formation in maxillary development. On the other hand, the palatal cleft was retained in the Ift88 fl/fl ;Wnt1Cre;p53 −/− mice, indicating that the excess bone formation or abnormal apoptosis was independent of the cleft palate phenotype in Ift88 mutant mice. Conclusions: Ift88 limits bone formation in the maxillary process by suppressing apoptosis.

Original languageEnglish
Pages (from-to)43-50
Number of pages8
JournalArchives of Oral Biology
Publication statusPublished - 1 May 2019


  • Apoptosis
  • Bone formation
  • Ift88
  • Maxillary process
  • Primary cilia


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