Spruijt Bart, Joosten Koen F M, Driessen Caroline, Rizopoulos Dimitris, Naus Nicole C, van der Schroeff Marc P, Wolvius Eppo B, van Veelen Marie-Lise C, Tasker Robert C, Mathijssen Irene M J
Rotterdam, The Netherlands; and Boston, Mass. From the Dutch Craniofacial Center; the Pediatric Intensive Care Unit; and the Departments of Biostatistics, Ophthalmology, Otorhinolaryngology, Oral and Maxillofacial Surgery, and Neurosurgery, Sophia Children's Hospital-Erasmus University Medical Center; and the Departments of Neurology and Anaesthesia (Pediatrics), Harvard Medical School and Boston Children's Hospital.
Plast Reconstr Surg. 2015 Aug;136(2):331-340. doi: 10.1097/PRS.0000000000001434.
The purpose of this study was to examine the relationship of head growth, obstructive sleep apnea, and intracranial hypertension in patients with syndromic or complex craniosynostosis, and to evaluate the authors' standardized treatment protocol for the management of intracranial hypertension in these patients.
The authors conducted a prospective observational cohort study of patients with syndromic craniosynostosis at a national referral center, treated according to a standardized protocol. Measurements included occipitofrontal head circumference, with growth arrest defined as downward deflection in occipitofrontal head circumference trajectory greater than or equal to a 0.5 SD fall from baseline over 2 years, or lack of change in occipitofrontal head circumference growth curve; sleep studies, with results dichotomized into no/mild versus moderate/severe obstructive sleep apnea; and funduscopy to indicate papilledema, supplemented by optical coherence tomography and/or intracranial pressure monitoring to identify intracranial hypertension.
The authors included 62 patients, of whom 21 (33.9 percent) had intracranial hypertension, 39 (62.9 percent) had obstructive sleep apnea, and 20 (32.3 percent) had occipitofrontal head circumference growth arrest during the study. Age at which intracranial hypertension first occurred was 2.0 years (range, 0.4 to 6.0 years). Preoperatively, 13 patients (21.0 percent) had intracranial hypertension, which was associated only with moderate/severe obstructive sleep apnea (p = 0.012). In the first year after surgery, intracranial hypertension was particularly related to occipitofrontal head circumference growth arrest (p = 0.006). Beyond 1 year after surgery, intracranial hypertension was associated with a combination of occipitofrontal head circumference growth arrest (p < 0.001) and moderate/severe obstructive sleep apnea (p = 0.007).
Children with syndromic craniosynostosis are at risk of intracranial hypertension. The major determinant of this after vault expansion is impaired head growth, which may occur at varying ages. The presence of moderate/severe obstructive sleep apnea also significantly increases the risk of intracranial hypertension.
CLINICIAL QUESTION/LEVEL OF EVIDENCE: Risk, III.
本研究旨在探讨综合征性或复杂性颅缝早闭患者的头部生长、阻塞性睡眠呼吸暂停和颅内高压之间的关系,并评估作者针对这些患者颅内高压管理的标准化治疗方案。
作者在一家国家级转诊中心对综合征性颅缝早闭患者进行了一项前瞻性观察队列研究,按照标准化方案进行治疗。测量指标包括枕额头围,生长停滞定义为枕额头围轨迹向下偏移,在2年内较基线下降大于或等于0.5个标准差,或枕额头围生长曲线无变化;睡眠研究,结果分为无/轻度与中度/重度阻塞性睡眠呼吸暂停;眼底镜检查以指示视乳头水肿,辅以光学相干断层扫描和/或颅内压监测以确定颅内高压。
作者纳入了62例患者,其中21例(33.9%)有颅内高压,39例(62.9%)有阻塞性睡眠呼吸暂停,20例(32.3%)在研究期间有枕额头围生长停滞。颅内高压首次出现的年龄为2.0岁(范围0.4至6.0岁)。术前,13例患者(21.0%)有颅内高压,这仅与中度/重度阻塞性睡眠呼吸暂停相关(p = 0.012)。术后第一年,颅内高压尤其与枕额头围生长停滞相关(p = 0.006)。术后1年以上,颅内高压与枕额头围生长停滞(p < 0.001)和中度/重度阻塞性睡眠呼吸暂停(p = 0.007)的联合情况相关。
综合征性颅缝早闭患儿有颅内高压风险。颅骨扩张后出现这种情况的主要决定因素是头部生长受损,这可能在不同年龄发生。中度/重度阻塞性睡眠呼吸暂停的存在也显著增加了颅内高压的风险。
临床问题/证据水平:风险,III级