Neurodevelopment in unilateral coronal craniosynostosis: a systematic review and meta-analysis

Annahita R. FotouhiDivision of Plastic and Reconstructive Surgery, Department of Surgery, Washington University School of Medicine, St. Louis, Missouri;

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Sarah N. ChiangDivision of Plastic and Reconstructive Surgery, Department of Surgery, Washington University School of Medicine, St. Louis, Missouri;

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Andrew M. PetersonDepartment of Otolaryngology–Head and Neck Surgery, Washington University School of Medicine, St. Louis, Missouri;

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Michelle M. DoeringBernard Becker Medical Library, Washington University School of Medicine, St. Louis, Missouri; and

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Gary B. SkolnickDivision of Plastic and Reconstructive Surgery, Department of Surgery, Washington University School of Medicine, St. Louis, Missouri;

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Sybill D. NaidooDivision of Plastic and Reconstructive Surgery, Department of Surgery, Washington University School of Medicine, St. Louis, Missouri;

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Jennifer M. StrahleDepartment of Neurosurgery, Washington University School of Medicine, St. Louis, Missouri

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Sean D. McEvoyDepartment of Neurosurgery, Washington University School of Medicine, St. Louis, Missouri

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Kamlesh B. PatelDivision of Plastic and Reconstructive Surgery, Department of Surgery, Washington University School of Medicine, St. Louis, Missouri;

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OBJECTIVE

The current literature on unilateral coronal craniosynostosis is replete with repair techniques and surgical outcomes; however, information regarding neurodevelopment remains unclear. Therefore, the aim of this systematic review and meta-analysis was to comprehensively assess the neurodevelopmental outcomes of patients with unicoronal craniosynostosis compared with their healthy peers or normative data.

METHODS

A systematic review of the Ovid MEDLINE, Embase, Web of Science, Scopus, Cochrane Library, and ClinicalTrials.gov databases from database inception to January 19, 2022, was performed. Included studies assessed neurodevelopment of patients with unicoronal craniosynostosis. Two independent reviewers selected studies and extracted data based on a priori inclusion and exclusion criteria. Results of developmental tests were compared with normative data or controls to generate Hedges’ g statistics for meta-analysis. The quality of included studies was evaluated using the National Institutes of Health Assessment Tool.

RESULTS

A total of 19 studies were included and analyzed, with an overall fair reporting quality. A meta-analysis of 325 postoperative patients demonstrated that scores of general neurodevelopment were below average but within one standard deviation of the norm (Hedges’ g = −0.68 [95% CI −0.90 to −0.45], p < 0.001). Similarly, postoperative patients exhibited lower scores in verbal, psychomotor, and mathematic outcome assessments.

CONCLUSIONS

This systematic review and meta-analysis found that patients with unicoronal craniosynostosis had poorer neurodevelopment, although scores generally remained within the normal range. These data may guide implementation of regular neurocognitive assessments and early learning support of patients with unicoronal craniosynostosis.

ABBREVIATIONS

PDI = Psychomotor Development Index; PI = prediction interval; WASI = Wechsler Abbreviated Scale of Intelligence; WISC = Wechsler Intelligence Scale for Children; WPPSI = Wechsler Preschool and Primary Scale of Intelligence.

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Figure from Candela-Cantó et al. (pp 61–70).

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