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Current Controversies in Craniosynostosis Research: A Tribute to Ian Jackson

T hroughout his career, Dr. Ian Jackson aimed to deepen our understanding of the innovations in which he was involved. He contributed a substantial body of research to the stabilizing and filling of bony defects of the cranial vault through the assessment of both inert bony substitutes, as well as through investigation of the osteogenic potential of bony and cellular elements. Our work in recent years has aimed to investigate controversial areas within craniosynostosis research, including the optimal timing and method of surgical intervention, the clinical diagnosis of certain craniosynostosis patterns, and developing the understanding of the consequences of severity of skull malformation on neurocognitive outcomes. Like the late Dr. Jackson, we aspire to pursue the lines of questioning that emerge from the data, wherever they may lead. Craniosynostosis, affecting 1 in 2000 to 2500 live births, is a condition in which one or more cranial sutures are prematurely fused. The resultant restriction on the skull leads to impaired calvarial growth perpendicular to the suture with a compensatory increase in growth at the remaining patent sutures. This pathology primarily affects early life, with the harmful consequences of restricted growth potentiated by the rapid development of the brain at this age. Beyond physical disfigurement, craniosynostosis has also been correlated with cognitive dysfunction. Though caused by external compression of the skull rather than an intrinsic defect to it, deformational plagiocephaly results in a similar cosmetic defect to craniosynostosis and is associated with possible negative cognitive effects as well. This review provides an update on research of the neurocognitive effects of this spectrum of disease as well as some future directions in this field.