Unusual case of carcinoma cuniculatum affecting the oral cavity of an 11-year-old boy.
Unusual case of carcinoma cuniculatum affecting the oral cavity of an 11-year-old boy.
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DOI：
10.1016/j.abd.2020.08.029
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发表时间：
2021-11
影响因子：
1.7
通讯作者：
Martinez R
中科院分区：
文献类型：
--
作者：
Niklander S;Sernuda LM;Martinez R
口腔楔状癌（OCC）是口腔鳞状细胞癌（OSCC）的一种罕见的分化良好的变体，于2005年首次纳入世界卫生组织（WHO）头颈部肿瘤分类。根据WHO的定义，OCC被定义为具有角蛋白核心和角蛋白填充的隐窝的广泛过程中的复层鳞状上皮的恶性肿瘤性增殖，其似乎钻入骨中，没有明显的恶性细胞学特征。1据估计，OCC占所有OSCC的2.7%。2迄今为止，报告的病例不到60例。在此我们报告一个罕见的病例，一个11岁的男孩因为左下唇的局部疼痛和感觉异常而就诊。他有II级室管膜瘤病史，通过手术和放疗进行了适当治疗，并进行了常规控制。他没有全身症状，已知的过敏或任何其他疾病的病史。口外检查未发现异常。口内，一个肿块，涉及颊和舌牙龈之间的左下第二磨牙和左下第一前磨牙观察。肿块的颊侧有一大片白色疣状斑片，同时在肿瘤的舌侧和颊侧也有囊性白色结节和浅表血管（图1）。肿块累及的牙齿是移动的。锥形束计算机断层扫描显示一个边界清晰的大透亮线，累及左下颌骨后部的整个厚度，贯穿颊侧骨板（图2）。切开活检显示肿瘤块由多个分支角蛋白填充的隐窝片组成，衬有表皮样上皮细胞。内生上皮细胞的生长模式深入牙槽骨，形成一个复杂的网络连接小管，类似于兔子的洞穴。上皮细胞分化良好，未见有丝分裂，无神经周浸润和淋巴管浸润。在间质的某些区域观察到主要由淋巴细胞组成的慢性炎性浸润，局部区域有中性粒细胞聚集（图3）。病变被诊断为口腔楔状癌。鉴别诊断
Oral Carcinoma Cuniculatum (OCC) corresponds to a rare, well-differentiated variant of Oral Squamous Cell Carcinoma (OSCC) first included in the World Health Organization (WHO) Classification of Head and Neck tumors in 2005. According to the WHO, OCC is defined as a malignant neoplastic proliferation of stratified squamous epithelium in broad processes with keratin cores and keratin-filled crypts which seem to burrow into the bone, without obvious cytological features of malignancy. 1 It is estimated that OCC accounts for up to 2.7% of all OSCC. 2 Up to date, less than 60 cases have been reported. Here we report an unsual case of an oral carcinoma cuniculatum affecting the gingiva of a child.An 11-year-old boy was consulted because of local pain and paresthesia of the left lower lip during an unknown amount of time. He had a history of grade II ependymoma, which was appropriately treated with surgery and radiotherapy and controlled routinely. He had no systemic symptoms, known allergies or medical history of any other illness. Extraoral examination revealed no anomalies. Intraorally, a mass involving both the buccal and lingual gingiva between the lower left second molar and lower left first premolar was observed. An extensive verrucous white patch was covering the buccal side of the mass, while cystic-like white nodules with superficial blood vessels were also present on both the lingual and buccal sides of the tumor (Fig. 1). The teeth involved in the mass were mobile. Cone-beam computed tomography revealed a well-defined large radiolucency involving the whole thickness of the posterior left mandible perforating the buccal bone plate (Fig. 2). An incisional biopsy was taken which showed a tumoral mass composed of multiple branching keratin-filled crypts sheets lined by epidermoid epithelial cells. The endophytic epithelial growth pattern burrowed deep inside into the alveolar bone, forming a complex network of connected canaliculi, resembling rabbit burrows. The epithelial cells were well differentiated, and no atypia, mitosis, perineural nor lymphovascular invasion was observed. A chronic inflamma tory infiltrates consisting mainly of lymphocytes with local areas of neutrophil accumulation was observed in some areas of the stroma (Fig. 3). The lesion was diagnosed as a carcinoma cuniculatum of the oral cavity. Differential diagnosis with
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影响因子：
3.3
作者：
Sun, Yan;Kuyama, Kayo;Yamamoto, Hirotsugu
通讯作者：
Yamamoto, Hirotsugu
DOI：
10.17796/jcpd.35.1.9qt8601640386651
发表时间：
2010-09-01
影响因子：
1.3
作者：
Hutton, A.;McKaig, S.;Parmar, S.
通讯作者：
Parmar, S.
DOI：
10.1016/j.oooo.2014.08.011
发表时间：
2014-12-01
影响因子：
2.9
作者：
Padilla, Ricardo J.;Murrah, Valerie A.
通讯作者：
Murrah, Valerie A.
DOI：
10.1002/hed.21493
发表时间：
2012-02-01
影响因子：
2.9
作者：
Pons, Yoann;Kerrary, Siham;Ruhin-Poncet, Blandine
通讯作者：
Ruhin-Poncet, Blandine