Outcome Measures Reported in Published Clinical Research Studies in Craniosynostosis: A Systematic Review.

ABSTRACT

CONTEXT The fair comparison of treatment interventions for craniosynostosis across different studies is expected to be impaired by incomplete reporting and the use of inconsistent outcomes. OBJECTIVE: This review assessed the outcomes currently reported in studies of craniosynostosis, and whether these outcomes are formally defined and prespecified in the study methods. DATA SOURCES, SEARCH TERMS, AND STUDY SELECTION Studies were sourced via an electronic, multi-database literature search for "craniosynostosis." All primary, interventional research studies published from 2011 to 2015 were reviewed. DATA EXTRACTION Two independent researchers assessed each study for inclusion and performed the data extraction. For each study, data were extracted on the individual outcomes reported, and whether these outcomes were defined and prespecified in the methods. DATA SYNTHESIS AND RESULTS: Of 1027 studies screened, 240 were included and proceeded to data extraction. These studies included 18,365 patients.2192 separate outcomes were reported. Of these, 851 outcomes (38.8%) were clearly defined, 1394 (63.6%) were prespecified in the study methods."Clinical and functional" was the most commonly reported outcome theme (900 outcomes, 41.1%), and "patient-reported" outcomes the least (7 outcomes, 0.3%)."Duration of surgery" was the most commonly reported single outcome (reported 80 times). "Cranial index" was the most variably defined outcome (18 different definitions used). CONCLUSION: The outcomes reported following treatment interventions for craniosynostosis are incompletely and variably defined. Improving definitions for these outcomes may aid comparison of different management strategies and improve craniosynostosis care. Suboptimal prespecification of these outcomes in the study methods implied that outcome reporting bias cannot be excluded.