The aim of this study was to test the hypothesis that obstructive sleep apnea syndrome (OSAS) exhibits oxidative stress and inflammation in patients who have a congenital, craniofacial anomaly.This prospective, cross-sectional cohort study included ambulant sleep study data to asses OSAS in patients with syndromic craniosynostosis and Treacher Collins syndrome. Laboratory analyses were performed including malondialdehyde, tumor necrosis factor α (TNF-α), interleukin 6, and high-sensitivity C-reactive protein.Forty-eight patients were included; 11 were adults; 37 were children. The patients' body mass indexes were normal, with a median (SD) of 0.7 (-1.82 to 2.48) in children and 20.5 (15.2-29.4) in adults. Obstructive sleep apnea syndrome was diagnosed in 23 of 48 patients. It was mild (median obstructive apnea-hypopnea index [oAHI], 2.3; oxygenation-desaturation index [ODI], 0.9) in 16 patients and moderate/severe in 7 patients (median oAHI, 10.8; ODI, 5.0). Neither oxidative stress nor inflammation had a correlation with the oAHI and ODI. Only TNF-α was found significantly higher in both the OSAS and non-OSAS groups compared with the reference values (median, 15.1 pg/mL and 12.3 pg/mL versus 4.05 [0.0-8.1 pg/mL], P < 0.001 and P < 0.001, respectively).Based on our findings we conclude that (mainly mild) OSAS, oxidative stress, as well as high-sensitivity C-reactive protein and interleukin 6 levels are not abnormal in the day time in a population of nonobese patients with a craniofacial anomaly. The increased level of TNF-α cannot be explained by OSAS. Future research should focus on mapping chronobiologic changes for further interpretation of the results.