Review
Copyright ©The Author(s) 2015. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Biol Chem. Aug 26, 2015; 6(3): 121-138
Published online Aug 26, 2015. doi: 10.4331/wjbc.v6.i3.121
Molecular basis of cleft palates in mice
Noriko Funato, Masataka Nakamura, Hiromi Yanagisawa
Noriko Funato, Masataka Nakamura, Research Center for Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo 113-8510, Japan
Hiromi Yanagisawa, Department of Molecular Biology, University of Texas Southwestern Medical Center, Dallas, TX 75390-9148, United States
Hiromi Yanagisawa, Life Science Center of Tsukuba Advanced Research Alliance, University of Tsukuba, Tsukuba, Ibaraki 305-8577, Japan
Author contributions: Funato N contributed to the conception and design of the manuscript, to the data analysis, and drafted the manuscript; Nakamura M and Yanagisawa H contributed to the interpretation of data, and revised the manuscript.
Supported by The Japan Society for the Promotion of Science (JSPS) through KAKENHI grants 25670774 and 15K11004, awarded to Funato N.
Conflict-of-interest statement: The authors declare no conflicts of interest.
Open-Access: This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/
Correspondence to: Noriko Funato, DDS, PhD, Research Center for Medical and Dental Sciences, Tokyo Medical and Dental University, 1-5-45 Yushima, Bunkyo-ku, Tokyo 113-8510, Japan. noriko-funato@umin.ac.jp
Telephone: +81-3-58035796 Fax: +81-3-58030203
Received: April 21, 2015
Peer-review started: April 22, 2015
First decision: May 13, 2015
Revised: May 26, 2015
Accepted: July 11, 2015
Article in press: July 14, 2015
Published online: August 26, 2015
Abstract

Cleft palate, including complete or incomplete cleft palates, soft palate clefts, and submucosal cleft palates, is the most frequent congenital craniofacial anomaly in humans. Multifactorial conditions, including genetic and environmental factors, induce the formation of cleft palates. The process of palatogenesis is temporospatially regulated by transcription factors, growth factors, extracellular matrix proteins, and membranous molecules; a single ablation of these molecules can result in a cleft palate in vivo. Studies on knockout mice were reviewed in order to identify genetic errors that lead to cleft palates. In this review, we systematically describe these mutant mice and discuss the molecular mechanisms of palatogenesis.

Keywords: Tbx1, Submucosal cleft palate, Incomplete cleft palate, Palatal shelf, Palatogenesis, Knockout mice

Core tip: Cleft lip and/or palate is one of the most frequent congenital craniofacial anomalies observed. Multifactorial conditions, including genetic and environmental factors, induce the formation of cleft palates. We screened knockout mice with cleft palate phenotypes and observed approximately 180 mice with the anomaly. In order to understand the molecular regulatory mechanisms of palatogenesis and to identify genetic errors that lead to cleft palates, we aimed to review studies performed using knockout mice with cleft palates.