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Experimental Animal Models in Cranial Suture Biology: Molecular and Pharmacological Treatment Strategies

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The Sutures of the Skull

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Abstract

The craniofacial skeleton develops both spatially and temporally, whereby cranial sutures are involved in regulating growth and morphogenesis. Disrupting these molecular and cellular interactions can result in craniosynostosis, the premature fusion of cranial sutures, which restricts the growth of the skull and causes malformations perpendicular to the sutures affected. Facial deformities and functional CNS irregularities can also occur. Expansion of the cranial vault and reconstructive surgery are still the main course of treatment, however they present an increased morbidity risk to the infant. Whilst the etiology of non-syndromic craniosynostosis has still not been decoded, the gain-of-function mutations in FGFR1-3 and TWIST1 are responsible for over three quarters of the most common craniofacial syndromes. Animal models have been extremely valuable in dissecting the role of genes in the cranial sutures and for developing other non-surgical treatments. In this review we present several pharmacological and molecule methods for treating craniosynostosis in animal models, which have been tested by using in vitro and in vivo assays; we then present a discussion of their possible use in humans with a focus on tyrosine kinase inhibitors.

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Authors and Affiliations

  1. Department of Plastic Surgery and Maxillofacial Surgery, Hôpital Universitaire Henri-Mondor, Créteil, France

    Martin Rachwalski

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Editors and Affiliations

  1. School of Medicine, Department of Neurosurgery, Aydın Adnan Menderes University, Efeler, Aydin, Turkey

    Mehmet Turgut

  2. Departments of Neurosurgery, Neurology, Surgery, and Structural and Cellular Biology, Tulane University School of Medicine, Center for Clinical Neurosciences, New Orleans, LA, USA

    R. Shane Tubbs

  3. Department of Radiology, Yüksek İhtisas University, Ankara, Turkey

    Ahmet T. Turgut

  4. Department of Neurosurgery, Tulane University, New Orleans, LA, USA

    Aaron S. Dumont

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Rachwalski, M. (2021). Experimental Animal Models in Cranial Suture Biology: Molecular and Pharmacological Treatment Strategies. In: Turgut, M., Tubbs, R.S., Turgut, A.T., Dumont, A.S. (eds) The Sutures of the Skull. Springer, Cham. https://doi.org/10.1007/978-3-030-72338-5_19

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