Wood Benjamin C, Oh Albert K, Keating Robert F, Boyajian Michael J, Myseros John S, Magge Suresh N, Rogers Gary F
Departments of 1 Plastic Surgery and.
Neurosurgery, Children's National Health System, Washington, DC.
J Neurosurg Pediatr. 2015 Sep;16(3):309-16. doi: 10.3171/2015.1.PEDS14464. Epub 2015 Jun 5.
OBJECT Progressive postnatal pansynostosis (PPP) is a rare form of craniosynostosis that is characterized by a normal head shape, insidious decrease in percentile head circumference, and high rates of elevated intracranial pressure (ICP). This investigation describes the clinical, radiographic, and genetic features of this entity. METHODS The authors' craniofacial database for the period 1997-2013 was retrospectively culled to identify patients who had a normal or near-normal head shape and CT-confirmed multiple-suture synostosis. Patients with kleeblatt-schädel or previous craniofacial surgery were excluded. All demographic information was collected and analyzed. RESULTS Seventeen patients fit the inclusion criteria. Nine patients had a syndromic diagnosis: Crouzon syndrome (n = 4), Pfeiffer syndrome (n = 2), Saethre-Chotzen syndrome (n = 1), Apert syndrome (n = 1), and achondroplasia (n = 1). With the exception of 3 patients with mild turricephaly, all patients had a relatively normal head shape. Patients were diagnosed at an average age of 62.9 months. Nearly all patients had some combination of clinical, radiographic, or ophthalmological evidence of increased ICP. CONCLUSIONS PPP is insidious; diagnosis is typically delayed because the clinical signs are subtle and appear gradually. All normocephalic infants or children with a known or suspected craniosynostotic disorder should be carefully monitored; any decrease in percentile head circumference or signs/symptoms of increased ICP should prompt CT evaluation.
目的 进行性产后全颅缝早闭(PPP)是一种罕见的颅缝早闭形式,其特征为头部形状正常、头围百分位数隐匿性减小以及颅内压(ICP)升高率高。本研究描述了该疾病的临床、影像学和遗传学特征。方法 回顾性筛选作者1997年至2013年期间的颅面数据库,以确定头部形状正常或接近正常且CT证实有多条颅缝早闭的患者。排除患有短头畸形或既往有颅面手术史的患者。收集并分析所有人口统计学信息。结果 17例患者符合纳入标准。9例患者有综合征诊断:克鲁宗综合征(n = 4)、费弗综合征(n = 2)、塞特雷-乔岑综合征(n = 1)、阿佩尔综合征(n = 1)和软骨发育不全(n = 1)。除3例有轻度塔头畸形的患者外,所有患者头部形状相对正常。患者的平均诊断年龄为62.9个月。几乎所有患者都有临床、影像学或眼科证据表明ICP升高的某种组合。结论 PPP隐匿性强;由于临床体征细微且逐渐出现,诊断通常延迟。所有头型正常的已知或疑似颅缝早闭疾病的婴儿或儿童都应仔细监测;头围百分位数的任何减小或ICP升高的体征/症状都应促使进行CT评估。