Need for standard outcome reporting systems in craniosynostosis

Abstract

Craniosynostosis is the premature fusion of one or more cranial sutures. When a cranial suture fuses prematurely, skull growth is altered and the head takes on a characteristic pathological shape determined by the suture(s) that fuses. Numerous treatment options have been proposed, but until recently there were no parameters or guidelines of care. Establishing such parameters was an important step forward in the treatment of patients with craniosynostosis, but results are still assessed using radiographic measurements, complication rates, and ad hoc reporting scales. Therefore, clinical outcome reporting in the treatment of craniosynostosis is inconsistent and lacks methodological rigor. Today, most reported evidence in the treatment of craniosynostosis is level 5 (expert opinion) or level 4 (case series) data. Challenges in obtaining higher quality level 1 or level 2 data include randomizing patients in a clinical trial as well as selecting the appropriate outcome measure for the trial. Therefore, determining core outcome sets that are important to both patients and health care professionals is an essential step in the evolution of caring for patients with craniosynostosis. Traditional clinical outcomes will remain important, but patient-reported outcomes, such as satisfaction, body image, functional results, and aesthetic outcomes, must also be incorporated if the selected outcomes are to be valuable to patients and families making decisions about treatment. In this article, the authors review the most commonly used tools to assess craniosynostosis outcomes and propose a list of longitudinal parameters of care that should be considered in the evaluation, diagnosis, and treatment evaluation of a patient with craniosynostosis.