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The State of Outcomes Research in Non-syndromic Craniosynostosis: A Systematic Review of the Literature over 20 Years
Liliana Camison, M.D., Joseph E. Losee, M.D., Jesse A. Goldstein, M.D.
University of Pittsburgh Medical Center
2014-03-15
Presenter: Liliana Camison
Affidavit:
I certify that Liliana, our research fellow, contributed a significant amount of effort to this project
Director Name: Joseph E. Losee, M.D.
Author Category: Fellow Plastic Surgery
Presentation Category: Clinical
Abstract Category: Craniomaxillofacial
Purpose: The field of craniofacial surgery was slow to embrace the outcomes movement. We aimed to examine the state of outcomes reporting in the non-syndromic craniosynostosis literature to identify strategies for improvement.
Methods: We conducted a systematic review of all English publications evaluating outcomes in non-syndromic craniosynostosis from 1993 to present. After applying pre-determined inclusion and exclusion criteria, study design, population specifics, and outcomes metrics were analyzed. Articles were rated according to three Level of Evidence scales. Four researchers independently reviewed all articles, with discrepancies resolved.
Results: 840 articles were identified. 135 met inclusion/exclusion criteria. There was a significant trend toward increasing annual rate of publications (R2=0.63). 38% of studies (n=52) analyzed patients with multiple forms of craniosynostosis compared to 31% (n=43) and 14% (n=19) on sagittal and metopic synostosis, respectively. Applying the ASPS level of evidence scale, 85% of articles were level 4, 10% level 3, only 4% level two, and none level 1. Average follow-up was 47 months (SD=36); 27.4% of studies did not state follow-up length. Outcome measures varied significantly between studies: 12% (n=17) used a validated outcomes measure and all 17 of these assessed neurodevelopment with established developmental scales. In contrast, 42% (n=57) of studies used an ad hoc measure for outcome analysis.
Conclusion: Most literature on non-syndromic craniosynostosis remains classified as a low level of evidence, calling for more rigorously designed trials. The large variation in reported outcomes for non-syndromic craniosynostosis validates the need for a more consistent approach to outcomes reporting in craniofacial surgery.
