Abstract
BackgroundAmeloblastomas are uncommon locally aggressive tumors of odontogenic epithelium that rarely metastasize. Currently, there is no standard of care for the metastatic forms. Several studies have shown that ameloblastomas frequently have a BRAF mutation.Case presentationWe report a case of a 33-year-old Caucasian woman with ameloblastoma diagnosed 30 years ago who developed lung metastasis 19 years ago. Systemic oral treatment with vemurafenib, a BRAF inhibitor, was initiated 28 months ago within the AcSé French basket clinical trial of vemurafenib.ConclusionsThe patient has shown a durable clinical, functional, and radiographic partial response with vemurafenib. These observations suggest the possibility of introducing neoadjuvant and/or adjuvant targeted therapy in locally advanced ameloblastoma to improve outcome. BRAF inhibition has proved to be an efficient strategy in patients with a BRAF-mutated ameloblastoma.
Highlights
Ameloblastomas are uncommon locally aggressive tumors of odontogenic epithelium that rarely metastasize
Ameloblastomas are rare odontogenic tumors of epithelial origin that are most frequently located in the posterior mandible
Surgical resection of the primary tumor with intent to cure is the mainstay of treatment for ameloblastomas and is considered the best approach to prevent recurrence and metastasis [3]
Summary
Cases of disseminated stage IV ameloblastoma are exceedingly rare, these observations suggest the possibility of introducing neoadjuvant and/or adjuvant targeted therapy in locally advanced ameloblastoma undergoing surgery to improve outcome and minimize functional and cosmetic morbidity.
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