Test-retest validation of a cranial deformity index in unilateral coronal craniosynostosis

Abstract

Unilateral coronal craniosynostosis (UCS) affects many infants resulting in abnormalities affecting the forehead and orbits. As a result, the deformity caused by UCS is very noticeable and there are several surgical treatment options available to normalize the head shape. However, there is a lack of consistently used outcome measures, resulting in difficulty assessing surgical outcomes and on-going debate over optimal treatments. Current techniques to quantify deformity in UCS are cumbersome, provide limited information, or are based on subjective assessments. In this study, a cranial deformity index was developed to quantify abnormality at the frontal bones for UCS that is accessible, user-friendly, and generates objective surface distance measurements. The cranial deformity index is defined as the Euclidean distance at the point of the largest deviation between the deformed skull compared to a reference skull. In addition, the index was successfully used to quantify post-operative changes in a single case of UCS that underwent corrective surgery. The reproducibility of the index was assessed using test-retest reliability and was demonstrated to be highly reproducible (ICC = 0.93). A user-friendly measurement index that is based on open-source software may be a valuable tool for surgical teams. In addition, this information can augment the consultation experience for patients and their families.

Keywords:

• Craniosynostosis
• unicoronal synostosis
• craniofacial
• pediatric
• surgery
• outcome measurement
• cranial vault remodelling
• 3D

Disclosure statement

No potential conflict of interest was reported by the author(s).

Additional information

Funding

This work was supposed by the Covenant Health Research Centre under Grant #13949.