Facial Suture Pathology in Syndromic Craniosynostosis: Human and Animal Studies.
Facial Suture Pathology in Syndromic Craniosynostosis: Human and Animal Studies.
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DOI：
10.1097/sap.0000000000002822
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发表时间：
2021-11-01
影响因子：
1.5
通讯作者：
Lee JC
中科院分区：
文献类型：
--
作者：
Wang MM;Haveles CS;Zukotynski BK;Reid RR;Lee JC
综合征性颅缝融合的面部畸形不仅在功能上、心理上和美学上受到损害，而且众所周知，重建起来也具有挑战性。在人类患者中，面部缝合融合是否在这些畸形的发病机制中起着重要作用的研究还不充分。使用按方法生成的检索词清单来查询MEDLINE数据库。文章收录由2名作者在独立评审后作出。文章提供了面部缝合参与综合征颅缝融合患者或疾病动物模型的洞察力。综合审查产生了19篇符合纳入标准的相关文章。20世纪中叶，颅面生物学家描述了面部缝合专利如何对正常的出生后面部发育至关重要。他们还提出，过早的骨化会扰乱生长载体，导致严重的畸形。最近，在综合征性颅缝融合的小鼠模型中，面部缝合融合被发现导致面中部畸形，而不受颅底病理的影响。最近很少有研究开始探索面部缝合对患者的影响，虽然它们为未来的研究铺平了道路，但它们存在明显的局限性。假设面部缝合融合与颅底病理共同作用，在综合征颅缝融合中产生显著的多灶性面部畸形，这可能从根本上改变手术治疗，并值得进一步研究。通过对文献进行系统的评估，这篇综述综合了到目前为止关于面部缝合在综合征颅缝融合中的作用的所有基础科学和人类临床研究，并阐明了未来研究的重要课题。我们最终确定了严格的影像研究的必要性，以纵向评估各种颅缝融合综合征患者的面部骨学。
Facial deformities in syndromic craniosynostosis are not only functionally, psychosocially, and aesthetically impairing but also notoriously challenging to reconstruct. Whether facial suture synostosis plays a significant role in the pathogenesis of these deformities is inadequately studied in human patients. The MEDLINE database was queried using a methodologically generated search term inventory. Article inclusion was adjudicated by 2 authors following independent review. Articles provided insight into facial suture involvement in either syndromic craniosynostosis patients or animal models of disease. Comprehensive review yielded 19 relevant articles meeting inclusion criteria. Mid-20th century craniofacial biologists characterized how patent facial sutures are essential for normal postnatal facial development. They also posited that premature ossification disrupts growth vectors, causing significant dysmorphologies. Recently, facial suture synostosis was found to cause midfacial deformities independent of cranial base pathology in mouse models of syndromic craniosynostosis. Few recent studies have begun exploring facial suture involvement in patients, and while they have paved the way for future research, they bear significant limitations. The hypothesis that facial suture synostosis acts in conjunction with cranial base pathology to produce the prominent, multifocal facial deformities in syndromic craniosynostosis may fundamentally alter surgical management and warrants further investigation. Methodically evaluating the literature, this review synthesizes all basic science and human clinical research thus far on the role of facial sutures in syndromic craniosynostosis and elucidates important topics for future research. We ultimately identify the need for rigorous imaging studies that longitudinally evaluate facial osteology across patients with various craniosynostosis syndromes.