Introduction: Fibrous Dysplasia (FD) is a fibro-osseous lesion of the osseous structures of the body. The exact etiology is not known. Monostotic (when one bone is involved), polyostotic (when multiple bones are involved) and craniofacial are the three subtypes of craniofacial dysplasia. When the lesions are limited to contiguous bones of the craniofacial skeleton, it is termed as craniofacial dysplasia. McCune Albright syndrome is the triad of polyostotic fibrous dysplasia, Cafe aulait spots and endocrine pathology. Case Report: This case describes craniofacial dysplasia in a 30 year old female patient who had unusual presentation on the right side of the face involving maxilla, sphenoid, ethmoid and orbit. The clinical features, radiological findings and treatment have been discussed.
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