Seruya Mitchel, Oh Albert K, Boyajian Michael J, Myseros John S, Yaun Amanda L, Keating Robert F, Rogers Gary F
Department of Plastic and Reconstructive Surgery, Children's National Medical Center, Washington, DC 20010, USA.
J Craniofac Surg. 2013 Jan;24(1):96-8. doi: 10.1097/SCS.0b013e318270fb83.
The severity and dysmorphology that results from the premature fusion of one or more cranial sutures is not uniform. Less striking phenotypes may be more easily missed on routine screening, possibly leading to delayed diagnosis and treatment. The purpose of this study was to compare the age at initial presentation for the different forms of craniosynostosis.
The authors reviewed the records of all patients who underwent open craniofacial repair of craniosynostosis at a single institution from 1996 to 2009. Relationships between type of suture fusion and age at initial consultation were compared.
Two hundred eleven patients (136 males, 75 females) were identified. Indications included sagittal (n = 96), metopic (n = 39), unicoronal (n = 33), bicoronal (n = 24), multisutural (n = 15), bilambdoidal (n = 3), and unilambdoidal (n = 1) synostoses. Seventeen patients (8.1%) had a craniosynostosis syndrome and 5 (2.4%) had a syndrome or disorder not typically associated with craniosynostosis [X-linked hypophosphatemic rickets (n = 3), achondroplasia (n = 1), and Beckwith Wiedemann (n = 1)]. Median age at initial consultation was 4.1 months; there was no gender difference. Patients with X-linked hypophosphatemic rickets presented at a significantly older age than nonsyndromic patients or those with a known craniosynostosis syndrome. Those with multisutural synostosis presented at a significantly older age than patients with sagittal or bicoronal synostosis.
Patients with multisutural involvement or X-linked hypophosphatemic rickets had a significant delay in presentation for craniosynostosis. The latter group of patients may especially benefit from routine surveillance for craniosynostosis given their advanced age at diagnosis.
一条或多条颅骨缝过早融合所导致的严重程度和形态异常并不一致。不太明显的表型在常规筛查中可能更容易被漏诊,这可能导致诊断和治疗延迟。本研究的目的是比较不同类型颅缝早闭的初次就诊年龄。
作者回顾了1996年至2009年在单一机构接受开放性颅面修复治疗颅缝早闭的所有患者的记录。比较了缝合融合类型与初次会诊年龄之间的关系。
共确定了211例患者(136例男性,75例女性)。适应证包括矢状缝(n = 96)、额缝(n = 39)、单冠状缝(n = 33)、双冠状缝(n = 24)、多缝(n = 15)、双侧人字缝(n = 3)和单侧人字缝(n = 1)早闭。17例患者(8.1%)患有颅缝早闭综合征,5例患者(2.4%)患有通常与颅缝早闭无关的综合征或疾病[X连锁低磷性佝偻病(n = 3)、软骨发育不全(n = 1)和贝克威思-维德曼综合征(n = 1)]。初次会诊的中位年龄为4.1个月;无性别差异。X连锁低磷性佝偻病患者的就诊年龄明显大于非综合征患者或患有已知颅缝早闭综合征的患者。多缝早闭患者的就诊年龄明显大于矢状缝或双冠状缝早闭患者。
多缝受累或X连锁低磷性佝偻病患者的颅缝早闭就诊明显延迟。鉴于后一组患者诊断时年龄较大,他们可能特别受益于颅缝早闭的常规监测。