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Neus Martínez-Abadías, Susan M. Motch, Talia L. Pankratz, Yingli Wang, Kristina Aldridge, Ethylin Wang Jabs and Joan T. Richtsmeier Tissue-specific responses to aberrant FGF signaling in complex head phenotypes Developmental Dynamics 242

Article first published online: 5 DEC 2012 | DOI: 10.1002/dvdy.23903

Key findings:

  • The most common Fgfr2 Crouzon syndrome mutation affects development of cranial tissues of varying embryological origin, as revealed by precise morphometric analysis of multi-modal images of heads of Fgfr2cC342Y/+ Crouzon syndrome mouse model.

  • Expanded catalogue of clinical craniofacial phenotypes in Crouzon syndrome caused by aberrant FGF/FGFR signaling includes skull, brain, nasopharynx, and eye dysmorphologies.

  • Effects of the Fgfr2c C342Y mutation on various head tissues points to primary effects of this mutation on cell–cell signaling required in the development of each of these tissues and their integration.

  • Findings contribute to the growing evidence that FGF/FGFR signaling is part of a multifaceted set of interactions among genes and regulatory networks that drive communication among cells and tissues in the development of the head that has been highly conserved in vertebrate evolution.

  • Multimodal imaging and 3D morphometric methods used to evaluate differences in tissue and organ development in Fgfr2cC342Y/+ Crouzon syndrome mice can be applied to other models of human disease.

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