Thyroid, Head and Neck Cancer (THANC) Foundation, New York, NY, USA.
Department of Otolaryngology-Head and Neck Surgery, Icahn School of Medicine at Mount Sinai, New York, NY, USA.
Ann Otol Rhinol Laryngol. 2022 Oct;131(10):1151-1157. doi: 10.1177/00034894211055591. Epub 2021 Oct 27.
Adenoid cystic carcinoma (ACC) is a commonly encountered salivary gland malignancy. However, it rarely occurs in the gingiva, an area generally thought to be devoid of minor salivary glands. We present a case occurring in this unusual site and review other reported cases.
A 56 year-old male presented with a right-sided mandibular toothache for 1 year and underwent dental extraction. Due to persistent pain, follow up examination revealed a large gingival lesion. A biopsy was positive for adenoid cystic carcinoma.
The patient underwent a complete right segmental mandibulectomy and was reconstructed with a fibular osteocutaneous free flap. Three months postoperatively, during the planning for adjuvant radiation therapy, the patient developed pain in the left mandible. Imaging revealed extensive involvement of the left native mandible. Deep bone biopsies in several areas of the left mandible revealed ACC. He then underwent a complete left hemi-mandibulectomy and reconstruction with a fibular osteocutaneous free flap. Tensor fascia lata suspension slings were placed due to concern for an open mouth deformity attributable to disruption of bilateral masticator slings. He will undergo adjuvant radiation therapy. Our review of the literature revealed 50 cases of gingival ACC published since 1972. Disease recurrence and distant metastases were noted in several patients, occurring at the latest after 30 years follow-up.
Given its indolent behavior, high proclivity for late recurrence and metastasis, and overall infrequency, ACC represents a pathology that requires early diagnosis and comprehensive long-term surveillance. While ACC is well described in oral cavity sites with high densities of minor salivary glands, it is not commonly seen in the gingiva. As such, gingival ACC may display a unique biological and/or clinical character. We offer the first literature review of this rare entity.
腺样囊性癌（ACC）是一种常见的涎腺恶性肿瘤。然而，它很少发生在牙龈，这个通常被认为没有小涎腺的区域。我们报告了一个发生在这个不寻常部位的病例，并回顾了其他报道的病例。
一名 56 岁男性因右侧下颌牙痛 1 年就诊，并接受了拔牙。由于持续疼痛，随访检查发现一个大的牙龈病变。活检显示为腺样囊性癌。
患者接受了右侧下颌骨节段切除术，并使用腓骨骨皮瓣进行重建。术后 3 个月，在计划辅助放疗时，患者出现左侧下颌骨疼痛。影像学显示左侧原生下颌骨广泛受累。左侧下颌骨多个部位的深部骨活检显示为 ACC。随后，他接受了左侧下颌骨半切除术，并使用腓骨骨皮瓣进行重建。由于担心因双侧咀嚼肌吊带断裂导致张口畸形，放置了阔筋膜张肌悬吊带。他将接受辅助放疗。我们对文献的回顾发现，自 1972 年以来，有 50 例牙龈 ACC 病例发表。一些患者出现疾病复发和远处转移，最晚在 30 年随访时发生。
鉴于其惰性行为、晚期复发和转移的高倾向以及总体罕见性，ACC 代表一种需要早期诊断和全面长期监测的病理学。虽然 ACC 在小涎腺密度高的口腔部位得到了很好的描述，但在牙龈中并不常见。因此，牙龈 ACC 可能表现出独特的生物学和/或临床特征。我们提供了对这种罕见实体的首次文献综述。
