From the Division of Plastic and Reconstructive Surgery, University of California, Los Angeles, CA.
Section of Plastic and Reconstructive Surgery, University of Chicago, Chicago, IL.
Ann Plast Surg. 2021 Nov 1;87(5):589-599. doi: 10.1097/SAP.0000000000002822.
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 after 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 although 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.
综合征性颅缝早闭患者的面部畸形不仅在功能、心理社会和审美方面造成损害,而且重建也极具挑战性。在人类患者中,面部缝线融合是否在这些畸形的发病机制中起重要作用,目前研究还不够充分。
使用方法学生成的搜索词库对 MEDLINE 数据库进行了查询。两位作者在独立审查后对文章进行了裁决。这些文章提供了关于综合征性颅缝早闭患者或疾病动物模型中面部缝线受累的见解。
全面审查产生了 19 篇符合纳入标准的相关文章。20 世纪中叶的颅面生物学家描述了开放的面部缝线对于正常的产后面部发育是多么重要。他们还假设,过早的骨化会破坏生长向量,导致明显的畸形。最近,在综合征性颅缝早闭的小鼠模型中发现,面部缝线融合会导致中面部畸形,而与颅底病变无关。最近的一些研究开始探索患者的面部缝线受累情况,尽管它们为未来的研究铺平了道路,但仍存在很大的局限性。
面部缝线融合与颅底病变共同作用,导致综合征性颅缝早闭中出现突出的多灶性面部畸形的假设可能从根本上改变手术管理,并值得进一步研究。通过系统地评估文献,本综述综合了迄今为止关于面部缝线在综合征性颅缝早闭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。我们最终确定需要进行严格的影像学研究,这些研究需要对各种颅缝早闭综合征患者的面部骨进行纵向评估。