From the Icahn School of Medicine at Mount Sinai, New York, NY.
Keck School of Medicine of the University of Southern California, Los Angeles, CA.
Ann Plast Surg. 2023 Jun 1;90(6S Suppl 5):S677-S680. doi: 10.1097/SAP.0000000000003496. Epub 2023 Mar 17.
Nonsyndromic unilateral coronal craniosynostosis (UCS) is a rare congenital disorder that results from premature fusion of either coronal suture. The result is growth restriction across the suture, between the ipsilateral frontal and parietal bones, leading to bony dysmorphogenesis affecting the calvarium, orbit, and skull base. Prior studies have reported associations between UCS and visual abnormalities. The present study utilizes a novel geometric morphometric analysis to compare dimensions of orbital foramina on synostotic versus nonsynostotic sides in patients with UCS. Computed tomography head scans of pediatric UCS patients were converted into 3-dimensional mesh models. Anatomical borders of left and right orbital structures were plotted by a single trained team member. Dimensions between synostotic and nonsynostotic sides were measured and compared. Medical records were examined to determine prevalence of visual abnormalities in this patient cohort. Visual abnormalities were reported in 22 of the 27 UCS patients (77.8%). Astigmatism (66.7%), anisometropic amblyopia (44.4%), and motor nerve palsies (33.3%) represented the 3 most prevalent ophthalmologic abnormalities. Orbits on synostotic sides were 11.3% narrower ( P < 0.001) with 21.2% less volume ( P = 0.028) than orbits on nonsynostotic sides. However, average widths, circumferences, and areas were similar between synostotic and nonsynostotic sides upon comparison of supraorbital foramina, infraorbital foramina, optic foramina, and foramina ovalia. Therefore, previously proposed compression or distortion of vital neurovascular structures within bony orbital foramina does not seem to be a likely etiology of visual abnormalities in UCS patients. Future studies will examine the role of ocular and/or neuro-ophthalmologic pathology in this disease process.
非综合征性单侧冠状缝早闭(UCS)是一种罕见的先天性疾病,源于冠状缝过早融合。结果是缝线处的生长受限,同侧额骨和顶骨之间,导致颅骨、眼眶和颅底的骨畸形形成。先前的研究报告了 UCS 与视觉异常之间的关联。本研究利用一种新的几何形态测量分析方法,比较 UCS 患者的颅缝早闭侧和非颅缝早闭侧眶孔的尺寸。将小儿 UCS 患者的计算机断层扫描头扫描转换为 3 维网格模型。由一名受过训练的团队成员绘制左眼和右眼眶结构的解剖边界。测量并比较颅缝早闭侧和非颅缝早闭侧之间的尺寸。检查病历以确定该患者队列中视觉异常的患病率。在 27 名 UCS 患者中,有 22 名(77.8%)报告存在视觉异常。散光(66.7%)、屈光不正性弱视(44.4%)和运动神经麻痹(33.3%)是最常见的 3 种眼科异常。颅缝早闭侧的眼眶狭窄 11.3%(P < 0.001),体积减少 21.2%(P = 0.028),小于非颅缝早闭侧。然而,比较眶上孔、眶下孔、视神经孔和卵圆孔后,颅缝早闭侧和非颅缝早闭侧的眶平均宽度、周长和面积相似。因此,以前提出的在骨性眶孔内重要的神经血管结构受压或变形似乎不是 UCS 患者视觉异常的可能病因。未来的研究将检查在这个疾病过程中眼和/或神经眼科病理学的作用。