Impact of surgery timing for craniosynostosis on neurodevelopmental outcomes: a systematic review

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There are currently no guidelines for the optimum age for surgical treatment of craniosynostosis. This systematic review summarizes and assesses evidence on whether there is an optimal age for surgery in terms of neurodevelopmental outcomes.


The databases MEDLINE, PsycINFO, CINAHL, Embase + Embase Classic, and Web of Science were searched between October and November 2016 and searches were repeated in July 2017. According to PICO (participants, intervention, comparison, outcome) criteria, studies were included that focused on: children diagnosed with nonsyndromic craniosynostosis, aged ≤ 5 years at time of surgery; corrective surgery for nonsyndromic craniosynostosis; comparison of age-at-surgery groups; and tests of cognitive and neurodevelopmental postoperative outcomes. Studies that did not compare age-at-surgery groups (e.g., those employing a correlational design alone) were excluded. Data were double-extracted by 2 authors using a modified version of the Cochrane data extraction form.


Ten studies met the specified criteria; 5 found a beneficial effect of earlier surgery, and 5 did not. No study found a beneficial effect of later surgery. No study collected data on length of anesthetic exposure and only 1 study collected data on sociodemographic factors.


It was difficult to draw firm conclusions from the results due to multiple confounding factors. There is some inconclusive evidence that earlier surgery is beneficial for patients with sagittal synostosis. The picture is even more mixed for other subtypes. There is no evidence that later surgery is beneficial. The authors recommend that future research use agreed-upon parameters for: age-at-surgery cut-offs, follow-up times, and outcome measures.

ABBREVIATIONS IQ = intelligence quotient; NSC = nonsyndromic craniosynostosis; QATSDD = Quality Assessment Tool for Reviewing Studies with Diverse Design; WASI = Wechsler Abbreviated Scale of Intelligence; WISC = Wechsler Intelligence Scale for Children; WRAT = Wide Range Achievement Test.

Article Information

Correspondence Rachel Mandela: Fielding House, St. James’s Hospital, Leeds, United Kingdom.

INCLUDE WHEN CITING Published online January 25, 2019; DOI: 10.3171/2018.10.PEDS18536.

Disclosures The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this paper.

© AANS, except where prohibited by US copyright law.



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    Flow diagram for identification of relevant studies (from 1990 to July 2017). Data added to the PRISMA template (from Moher D, Liberati A, Tetzlaff J, Altman DG, The PRISMA Group (2009). Preferred Reporting Items for Systematic Reviews and Meta-Analyses: The PRISMA Statement. PLoS Med 6(7): e1000097) under the terms of the Creative Commons Attribution License. Figure is available in color online only.

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    Risk of bias summary for individual studies: review authors’ judgments about each risk of bias item for each included study. A minus sign (−) indicates a high risk of bias; a plus sign (+), a low risk; and a question mark (?), an unclear risk. Figure is available in color online only.

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    Risk of bias graph: review authors’ judgments about each risk of bias item presented as percentages across all included studies. Figure is available in color online only.



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