Children with craniosynostosis are at risk for increased intracranial pressure, and additional possibilities to screen for increased intracranial pressure are required. The authors' aim was to use ultrasound measurements of the optic nerve sheath to understand and express the variability of intracranial pressure in syndromic craniosynostosis. Therefore, five pediatric patients with craniosynostosis underwent invasive 24-hour intracranial pressure monitoring and simultaneous optic nerve sheath measurements. In three patients, the intracranial pressure was abnormal, and during the second half of the night, the optic nerve sheath was increased in all three patients. The optic nerve sheath diameter changes during the night and is as dynamic as the intracranial pressure. To the best of their knowledge, the authors are the first to describe a real-time relationship of the optic nerve sheath with increased intracranial pressure in children.