The purpose of this study was to investigate the effects of early spheno-occipital synchondrosis (SOS) fusion in preadolescent patients with syndromic craniosynostosis (SC) on the craniofacial skeletal patterns. Twenty preadolescent SC patients were divided into the fused SOS (FS, n ¼ 10; 8 Crouzon and 2 Apert) and not-fused SOS groups (NFS, n ¼ 10; 9 Crouzon and 1 Apert). Lateral cephalograms (mean age: 9.60 years, cervical vertebral maturation index: stage I and II) were used to investigate the skeletal sagittal (ANB) and vertical patterns (SN-GoMe), upward inclination of the anterior cranial base (ACB; SN-FH), degree of midface hypoplasia (MH, SNA), retrusive position of orbitale (SNO), and forward position of the condyle in relation to sella (saddle angle). Using the ordinal values calculated by ethnic norm (criteria: moderate, over AE1 standard deviation, severe, over AE2 standard deviation), statistical analysis was performed. The FS group showed a higher percentage of severe MH than the NFS group (70% versus 10%, P < 0.05). Although the 2 groups did not differ in the distribution of ANB, SN-GoMe, saddle angle, and SN-FH (all P > 0.05), the FS group showed relatively higher percentages of severe Class III (100% versus 70%), severe hyper-divergent pattern (40% versus 10%), severely forward condyle position (30% versus 0%), and moderate and severe upward anterior cranial base inclination (90% versus 50%) than the NFS group. However, the 2 groups exhibited the same distribution of moderately and severely retrusive orbitale position ([50%, 20%], P > 0.05). Early SOS fusion in preadolescent SC patients might not be related to retrusive orbitale position, but to severe MH.