Kim Jong-Ho, Kang Jiwon, Kim Seong-Ik, Kim Byung Jun
Department of Plastic and Reconstructive Surgery, Seoul National University College of Medicine, Seoul National University Bundang Hospital, Republic of Korea.
Department of Pathology, Seoul National University College of Medicine, Seoul, Republic of Korea.
Arch Plast Surg. 2023 Feb 6;50(1):49-53. doi: 10.1055/s-0042-1758468. eCollection 2023 Jan.
Juvenile ossifying fibroma (JOF) is a variant of the ossifying fibroma and includes two histopathological subtypes: trabecular and psammomatoid. Psammomatoid JOF (PJOF) in craniofacial structures should be distinguished from other fibro-osseous lesions, such as fibrous dysplasia (FD), considering the difference in the treatment protocols. Here, we present a rare case of PJOF that was initially misdiagnosed as a case of FD and emphasize the importance of considering JOF in the differential diagnosis of patients with craniofacial fibro-osseous lesions. A 4-year-old boy demonstrated progressive enlargement of the zygomaticomaxillary area on his left side for the last 6 months. The patient was diagnosed as a case of FD based on the clinical features and radiographic findings, and was operated considering the rapid progression. To achieve facial symmetry, contouring of the zygomatic bone and arch was performed. However, the patient demonstrated rapid enlargement at the 3-month postoperative follow-up. The decision was made to surgically remove the tumor due to visual field impairment. Intraoperatively, a rubbery mass, which was separated from the surrounding cortical bone, was identified and excised. The lesion was confirmed as PJOF by histopathological examination. The possibility of PJOF should not be ruled out in the differential diagnosis of patients with fibrous-osseous lesions. In the event of suspected PJOF, accurate diagnosis should be made through definitive biopsy.
青少年骨化性纤维瘤（JOF）是骨化性纤维瘤的一种变体，包括两种组织病理学亚型：小梁型和砂粒体型。考虑到治疗方案的差异，颅面结构中的砂粒体型青少年骨化性纤维瘤（PJOF）应与其他纤维骨性病变，如骨纤维异常增殖症（FD）相鉴别。在此，我们报告一例罕见的PJOF病例，该病例最初被误诊为FD，并强调在颅面纤维骨性病变患者的鉴别诊断中考虑JOF的重要性。一名4岁男孩在过去6个月中左侧颧上颌区域逐渐肿大。根据临床特征和影像学表现，该患者被诊断为FD，并因病情快速进展而接受手术。为实现面部对称，对颧骨和颧弓进行了轮廓修整。然而，患者在术后3个月的随访中出现快速肿大。由于视野受损，决定手术切除肿瘤。术中，发现并切除了一个与周围皮质骨分离的橡胶样肿块。经组织病理学检查，该病变被确认为PJOF。在纤维骨性病变患者的鉴别诊断中，不应排除PJOF的可能性。如果怀疑是PJOF，应通过明确的活检进行准确诊断。
