Glandular odontogenic cyst (GOC) is a rare jawbone cyst of odontogenic origin. Human patched (PTCH) is a tumour suppressor gene that has been recently associated with signalling pathways during odontogenesis. Recently alterations of this gene were found on sporadic odontogenic keratocysts. This evidence, together with the biological behaviour similarities of both lesions, and the absence of reports on molecular analysis of GOC, led us to hypothesize that PTCH gene mutations may underlie the tumorigenesis of GOC. Therefore the aim of this study was to report one additional case of GOC and investigate the PTCH gene of the cyst. No mutations were found in the splicing and coding regions of the PTCH gene. In conclusion, the PTCH gene does not seem to be involved in GOC pathogenesis.