成釉细胞纤维牙瘤(AFO)是一种相对罕见的,良性非侵入性混合牙源性肿瘤来源于牙组织的上皮和外胚间充质成分。它的平均年龄通常为11.5岁,位于下颌骨的后段。在上颌骨后部极为罕见。尽管最新的世卫组织版本将AFO分类为正在发展的牙瘤,在这里,我们介绍了一名21岁男性的局部侵袭性AFO,涉及上颌骨后部和鼻窦并伴有骨破坏。患者有两年的缓慢进行性左侧面部肿胀并伴有恶臭引流的病史。CT扫描显示5.5x4.3cm界限清楚的可膨胀肿块,混合衰减和周围钙化占据了左上颌骨和窦,硬腭和眶缘的骨破坏。根据文献,大多数AFO病例都通过保守方法进行了充分治疗,只需摘除或手术刮除。据我们所知,我们的病例是第一例积极接受左上颌骨切除术的病例,腭切除术,和重建手术,因为它的放射学发现,这表明是局部浸润性肿瘤。组织学上,标本显示了增生性上皮的混合物,间充质组织元件,以及由牙釉质基质和牙质沉积物组成的可变量的矿化沉积物,最终诊断为AFO。总之,我们提出了一个罕见的AFO病例,具有不寻常的侵略性表现,年龄组,和网站参与。射线照相,组织病理学特征,这种不寻常的局部侵袭性肿瘤的治疗方法和相关文献的综述。
Ameloblastic fibro-odontoma (AFO) is a relatively rare, benign noninvasive mixed odontogenic neoplasm derived from epithelial and ectomesenchymal elements of the dental tissues. It usually presents with a mean age of 11.5 years and in the posterior segment of the mandible. It is extremely rare in the posterior maxilla. Although the latest WHO edition classified AFO as developing odontoma, here we present a locally aggressive AFO in a 21-year-old male involving the posterior maxilla and sinus with bone destruction. The patient presents with a two-year history of slowly progressive left facial swelling with malodorous drainage. The CT scan revealed a 5.5 x 4.3 cm well-circumscribed expansile mass with mixed attenuation and peripheral calcification occupying the left maxilla and sinus with bone destruction of the hard palate and orbital rim. According to the literature, most of the AFO cases were treated adequately through a conservative approach with just enucleation or surgical curettage. To our knowledge, our
case is the first
case treated aggressively with left maxillectomy, palatectomy, and reconstruction surgery because of its radiologic findings, which suggested a locally invasive neoplasm. Histologically, the specimen showed a mixture of proliferative epithelial, mesenchymal tissue elements, and variable amounts of mineralized deposits consisting of enamel matrix and dentinoid deposits, and the final diagnosis was AFO. In conclusion, we present a rare
case of AFO with an unusual aggressive presentation, age group, and site involved. The radiographic, histopathologic features, and therapeutic approaches of this unusual locally aggressive tumor are presented with the review of relevant literature.