Department of Oral Diagnosis, Piracicaba Dental School, University of Campinas (UNICAMP), Av. Limeira, 901, Piracicaba, São Paulo, CEP: 13414-903, Brazil.
Dental Oncology Service, Instituto do Câncer do Estado de São Paulo, ICESP-FMUSP, São Paulo, Brazil.
Head Neck Pathol. 2021 Sep;15(3):757-768. doi: 10.1007/s12105-020-01277-2. Epub 2021 Jan 4.
Neuroblastoma is the most common extracranial solid cancer of infancy, occurring mainly in the adrenal gland, with high metastatic potential. However, involvement of the head and neck region is rare. Here, we present two cases of metastatic neuroblastoma of childhood, in which a mandibular swelling was the first sign of disseminated disease. Case 1 describes a 4-year-old boy with a 2-week history of painful swelling in the left mandibular region, body soreness and weakness. Panoramic radiography and computed tomography showed a destructive lesion in the left mandibular ramus. Case 2 describes a 3-year-old boy with a 1-month history of swelling in the right mandibular area. Panoramic radiograph and cone-beam computed tomography showed a destructive lesion in the right body and ramus of the mandible, displacing tooth germs, with the destruction of vestibular and lingual bone cortices. In both cases, microscopic analyses revealed a diffuse proliferation of small, round, and blue cells with hyperchromatic nuclei and scant cytoplasm. While Case 1 was more undifferentiated, Case 2 presented eosinophilic areas suggestive of neuropil. A large immunohistochemical panel was performed, showing expression of neural markers such as CD56, neuron-specific enolase (in Case 2), chromogranin, and synaptophysin. Both lesions presented a high proliferation index (Ki67 > 70% and 80%, respectively). Positron emission tomography-computed tomography revealed ipsilateral adrenal primary lesions in both cases, with multiple bone metastatic lesions. Besides the mandible, multiple sites of the axial and appendicular skeleton were affected. Treatment consisted of induction chemotherapy, adrenalectomy, consolidation chemoradiotherapy, and post-consolidation therapy.
神经母细胞瘤是婴儿期最常见的颅外实体癌,主要发生于肾上腺,具有较高的转移潜能。然而,头颈部受累较为罕见。在此,我们报告两例儿童转移性神经母细胞瘤病例,下颌肿胀是播散性疾病的首发征象。病例 1 描述了 1 例 4 岁男孩,2 周前出现左侧下颌区域疼痛性肿胀、全身酸痛和乏力。全景片和 CT 显示左侧下颌支破坏性病变。病例 2 描述了 1 例 3 岁男孩,1 个月来出现右侧下颌区域肿胀。全景片和锥形束 CT 显示右侧下颌体和支破坏性病变,推移牙胚,伴有颊侧和舌侧骨皮质破坏。在这两个病例中,显微镜分析显示小而圆的蓝色细胞弥漫性增生,细胞核深染,细胞质稀少。病例 1 分化程度较低,病例 2 呈现嗜酸性区,提示神经毡。进行了广泛的免疫组织化学检测,显示神经标志物如 CD56、神经元特异性烯醇化酶(病例 2)、嗜铬粒蛋白和突触素的表达。两个病变均具有较高的增殖指数(Ki67 分别为>70%和 80%)。正电子发射断层扫描-CT 显示两个病例均存在同侧肾上腺原发灶,伴有多发骨转移灶。除下颌骨外,还累及多个轴性和附肢骨骼部位。治疗包括诱导化疗、肾上腺切除术、巩固化疗和放疗,以及巩固治疗后。