Kevin Bloch-Maier, Sophie-Myriam Dridi, Arek Sulukdjian, Anne-Laure Ejeil
Department of Maxillofacial Surgery, Beaujon hospital, AP-HP, Paris, France.
Department of Periodontology, Institute of Oral Medicine Riquier, CHU Nice, Nice, France.
Orphanet J Rare Dis. 2025 Sep 24;20(1):488. doi: 10.1186/s13023-025-04010-w.
Xanthogranulomatous inflammation is a rare chronic inflammatory condition. It has been described in the long bones, and can affect various organs, including the salivary glands, gallbladder, kidneys, and gastrointestinal tract. The diagnosis is made throught histopathology which reveales foamy macrophages alongside polymorphonuclear leukocytes, plasma cells, and polyclonal lymphocytes arranged in a mosaic-like pattern. Here, we present the case of a 16-year-old boy in good general health who was referred by his orthodontist to the oral surgery department at Bretonneau Hospital for an asymptomatic multipartite image of the left mandibular angle that was discovered incidentally on a panoramic radiograph. The clinical examination was unremarkable. Cone beam computed tomography revealed a multipartite osteolytic lesion of the left mandibular angle, but this that was not specific enough for a diagnosis. Magnetic resonance imaging ruled out a vascular malformation. A biopsy revealed xanthogranulomatous osteomyelitis. In summary, this is the second case of xanthogranulomatous osteomyelitis localised to the mandible. This highlights a crucial point : radiological images of xanthogranulomatous osteomyelitis do not allow for a diagnosis; a biopsy is essential to rule out an aggressive or a malignant tumor.
黄色肉芽肿性炎症是一种罕见的慢性炎症性疾病。它已在长骨中被描述，可累及包括唾液腺、胆囊、肾脏和胃肠道在内的各种器官。通过组织病理学进行诊断，组织病理学显示泡沫状巨噬细胞与多形核白细胞、浆细胞和多克隆淋巴细胞呈镶嵌样排列。在此，我们报告一例16岁身体健康的男孩，他被正畸医生转诊至布雷托诺医院口腔外科，原因是在全景X线片上偶然发现左下颌角无症状的多部分影像。临床检查无异常。锥形束计算机断层扫描显示左下颌角有一个多部分溶骨性病变，但这不足以明确诊断。磁共振成像排除了血管畸形。活检显示为黄色肉芽肿性骨髓炎。总之，这是第二例局限于下颌骨的黄色肉芽肿性骨髓炎病例。这突出了一个关键点：黄色肉芽肿性骨髓炎的放射学图像无法确诊；活检对于排除侵袭性或恶性肿瘤至关重要。
