Odontogenic myxoma is a rare benign neoplasm with locally aggressivebehaviour and a high risk of recurrence. It has a predilection for the man-dible and occurs in the 2nd–3rd decade. The authors report a case of anodontogenic myxoma presenting with a spontaneous oro-nasal fistula in a29-year-old male. Histo-pathological analysis of the incisional biopsyspecimen initially suggested a diagnosis of calcifying epithelial odontogenictumour. A partial maxillectomy was performed, and following assessmentof the resection specimen, a diagnosis of odontogenic myxoma was ren-dered. A 3 cm bicortical iliac crest bone graft was harvested and inset to theright maxillary defect under general anaesthesia. Rehabilitation will becompletedwiththeplacementofimplants. Casereport A 29-year-old non-Caucasian male of Chinese descentwas referred by his general dental practitioner to hislocal oral and maxillofacial surgery department forassessmentofaspontaneouspalatalfistulaintheupperright maxilla. His medical history was unremarkable.He was a non-smoker and did not consume alcohol.Intra-oral examination revealed an oro-nasal fistula inthe upper right, UR3-4 region. There was no palpablecervicallymphadenopathy.Plain radiographs including an Orthopantomograph(Fig. 1), a periapical (Fig. 2) and an upper anteriorocclusal demonstrated a radiolucency in this region.Further investigation with a cone-beam computedtomography(CT)revealedaradiolucencyintheUR3-4region, resorbing the palatal cortical bone, alveolarridge and the roots of UR3-4. An incisional biopsy wasperformed under local anaesthesia via a palatal flap.Histopathological investigation identified nests andsmall aggregates of polyhedral epithelial cells indegenerated fibro-collagenous stroma with slightmyxoid degeneration, accompanied by amyloiddeposits. Special staining with Congo red was positivein these deposits. The immunohistochemical stainMNF116 decorated the epithelial cells. Ki67 prolifera-tive index was low. A diagnosis of calcifying epithelialodontogenic tumour was made by clinico-pathologiccorrelation.The surgical management involved a partialmaxillectomy performed under general anaesthesia. Amucoperiosteal flap was raised via a crevicular incisionfrom UR6 to UR2, and no bony fenestration wasapparent. UR2–UR6 were extracted, and the partialmaxillectomy was completed with an extra marginsuperiorly excised to ensure clearance. Histo-pathological assessment was inconclusive, and sevenslides were sent for a second opinion to Leeds TeachingHospital. No evidence of calcifying epithelialodontogenictumourwasfoundinthesamples.Severalsections showed inactive-looking odontogenic epithe-lium (Fig. 3), lying within myxoid fibrous connectivetissue (Fig. 4). There was clear evidence of localdestruction of bone (Fig. 5) and tooth-root in areas.