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Mechanical loading of cranial joints minimizes the craniofacial phenotype in Crouzon syndrome

Children with syndromic forms of craniosynostosis undergo a plethora of surgical interventions to resolve the clinical features caused by the premature fusion of cranial sutures. While surgical correction is reliable, the need for repeated rounds of invasive treatment puts a heavy burden on the chil...

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Autores principales: Moazen, Mehran, Hejazi, Mahbubeh, Savery, Dawn, Jones, Dominic, Marghoub, Arsalan, Alazmani, Ali, Pauws, Erwin
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Nature Publishing Group UK 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9188582/
https://www.ncbi.nlm.nih.gov/pubmed/35690633
http://dx.doi.org/10.1038/s41598-022-13807-9
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author Moazen, Mehran
Hejazi, Mahbubeh
Savery, Dawn
Jones, Dominic
Marghoub, Arsalan
Alazmani, Ali
Pauws, Erwin
author_facet Moazen, Mehran
Hejazi, Mahbubeh
Savery, Dawn
Jones, Dominic
Marghoub, Arsalan
Alazmani, Ali
Pauws, Erwin
author_sort Moazen, Mehran
collection PubMed
description Children with syndromic forms of craniosynostosis undergo a plethora of surgical interventions to resolve the clinical features caused by the premature fusion of cranial sutures. While surgical correction is reliable, the need for repeated rounds of invasive treatment puts a heavy burden on the child and their family. This study explores a non-surgical alternative using mechanical loading of the cranial joints to prevent or delay craniofacial phenotypes associated with Crouzon syndrome. We treated Crouzon syndrome mice before the onset of craniosynostosis by cyclical mechanical loading of cranial joints using a custom designed set-up. Cranial loading applied to the frontal bone partially restores normal skull morphology, significantly reducing the typical brachycephalic appearance. This is underpinned by the delayed closure of the coronal suture and of the intersphenoidal synchondrosis. This study provides a novel treatment alternative for syndromic craniosynostosis which has the potential to be an important step towards replacing, reducing or refining the surgical treatment of all craniosynostosis patients.
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spelling pubmed-91885822022-06-13 Mechanical loading of cranial joints minimizes the craniofacial phenotype in Crouzon syndrome Moazen, Mehran Hejazi, Mahbubeh Savery, Dawn Jones, Dominic Marghoub, Arsalan Alazmani, Ali Pauws, Erwin Sci Rep Article Children with syndromic forms of craniosynostosis undergo a plethora of surgical interventions to resolve the clinical features caused by the premature fusion of cranial sutures. While surgical correction is reliable, the need for repeated rounds of invasive treatment puts a heavy burden on the child and their family. This study explores a non-surgical alternative using mechanical loading of the cranial joints to prevent or delay craniofacial phenotypes associated with Crouzon syndrome. We treated Crouzon syndrome mice before the onset of craniosynostosis by cyclical mechanical loading of cranial joints using a custom designed set-up. Cranial loading applied to the frontal bone partially restores normal skull morphology, significantly reducing the typical brachycephalic appearance. This is underpinned by the delayed closure of the coronal suture and of the intersphenoidal synchondrosis. This study provides a novel treatment alternative for syndromic craniosynostosis which has the potential to be an important step towards replacing, reducing or refining the surgical treatment of all craniosynostosis patients. Nature Publishing Group UK 2022-06-11 /pmc/articles/PMC9188582/ /pubmed/35690633 http://dx.doi.org/10.1038/s41598-022-13807-9 Text en © The Author(s) 2022 https://creativecommons.org/licenses/by/4.0/Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) .
spellingShingle Article
Moazen, Mehran
Hejazi, Mahbubeh
Savery, Dawn
Jones, Dominic
Marghoub, Arsalan
Alazmani, Ali
Pauws, Erwin
Mechanical loading of cranial joints minimizes the craniofacial phenotype in Crouzon syndrome
title Mechanical loading of cranial joints minimizes the craniofacial phenotype in Crouzon syndrome
title_full Mechanical loading of cranial joints minimizes the craniofacial phenotype in Crouzon syndrome
title_fullStr Mechanical loading of cranial joints minimizes the craniofacial phenotype in Crouzon syndrome
title_full_unstemmed Mechanical loading of cranial joints minimizes the craniofacial phenotype in Crouzon syndrome
title_short Mechanical loading of cranial joints minimizes the craniofacial phenotype in Crouzon syndrome
title_sort mechanical loading of cranial joints minimizes the craniofacial phenotype in crouzon syndrome
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9188582/
https://www.ncbi.nlm.nih.gov/pubmed/35690633
http://dx.doi.org/10.1038/s41598-022-13807-9
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