Objective: To gain more information on the oral health status of subjects with syndromic craniosynostosis. Design: A cross-sectional study. Materials and Methods: The present study took place at the Hospital for Rehabilitation of Craniofacial Anomalies of University of São Paulo (HRAC-USP). The sample was 19 patients with syndromic craniosynostosis (10 Apert, 5 Crouzon, 2 Pfeiffer and 2 Saethre-Chotzen), aged 5 to 15 years. An assessment of plaque, caries and gingival indices, and evaluation of the efficacy of toothbrushing was carried out. The measurements included PHP index, dmft and DMFT indices, gingival index, comparison of PHP before and after non-supervised toothbrushing and between individuals with and without severe syndactyly. Results: The patients displayed high plaque index and poor efficacy of toothbrushing, regardless of the presence of severe syndactyly; despite the plaque accumulation, the gingival index was not proportionally high. There was predominance of the D component for the DMFT index, which combined with the need for restorative treatment in 42.1% of the patients indicates poor access to dental care by these patients. Conclusions: The results show the need for a dental follow-up programme for these patients. Carers should be informed of the importance in aiding these patients during accomplishment of oral hygiene at home. Keywords: acrocephalosyndactylia, craniofacial dysostosis, craniosynostosis dental plaque index, DMF index, periodontal index, mouth breathing, toothbrushing