Kitabata Reina, Sakamoto Yoshiaki, Miwa Tomoru, Yoshida Kazunari, Kishi Kazuo
Department of Plastic and Reconstructive Surgery, Keio University School of Medicine.
Department of Neurosurgery, Keio University School of Medicine, Tokyo, Japan.
J Craniofac Surg. 2020 Jul-Aug;31(5):e475-e477. doi: 10.1097/SCS.0000000000006441.
Sagittal craniosynostosis is the most common of all craniosynostoses. Patients with sagittal craniosynostosis exhibit a typical cranial appearance, including scaphocephaly, and an elongated head, with a fused and ridged sagittal suture. Moreover, some recent reports described atypical sagittal craniosynostosis accompanied by autism, speech delay, and hyperactivity. This condition, known as delayed-onset craniosynostosis, is rarely reported, given that it is difficult to determine if the condition is congenital or has a delayed-onset.This report describes the clinical course and treatment of 2 brothers with atypical sagittal synostosis. The shapes of their heads were not indicative of scaphocephaly and the younger brother exhibited delayed-onset sagittal synostosis. Their father and paternal grandmother exhibited similar cranial morphologies. Therefore, we hypothesized the involvement of a familial factor in the etiology of atypical sagittal synostosis in these patients.
矢状缝早闭是所有颅缝早闭中最常见的。矢状缝早闭患者表现出典型的颅骨外观,包括舟状头畸形和头部拉长,矢状缝融合并隆起。此外,最近一些报告描述了伴有自闭症、语言发育迟缓及多动的非典型矢状缝早闭。这种情况被称为迟发性颅缝早闭,鉴于难以确定该病症是先天性的还是迟发性的,故鲜有报道。本报告描述了2例患有非典型矢状缝早闭的兄弟的临床病程及治疗情况。他们的头部形状并不提示舟状头畸形,且弟弟表现为迟发性矢状缝早闭。他们的父亲和祖母表现出相似的颅骨形态。因此,我们推测家族因素参与了这些患者非典型矢状缝早闭的病因。