Background: Fibrous dysplasia (FD) of bone is a rare sporadic benign congenital condition in which normal bone is replaced by fibro-osseous tissue with immature osteogenesis. Sarcomatous transformation is exceptional. The most common presenting features in craniofacial forms include atypical facial pain and headaches. Case Presentations: we report here 2 cases, that of a 36 year old male and of a 43 female, who both presented with a similar history of headaches. The male patient refused surgery so removal of the diseased bone was not an option. However, as the limited growth of this lesion have been established, only further, albeit close, observation was planned. The female patient underwent a successful removal of her tumour through an endoscopic approach. At the 1.5 year follow-up, she remains disease free. Conclusion: craniofacial fibrous dysplasia can present itself in myriad ways, therefore, the diagnosis should always be considered in case of headache, neuralgia or sensory disorders. Modern imaging modalities and histopathologic analysis have made the diagnosis reasonably straightforward. A medico-surgical approach is useful for these patients. Surgery ―when indicated― is tailored to the specific clinical presentation. The goal is to be as minimally invasive as possible while still achieving the desired outcome, prioritizing the preservation of existing function.