Thompson Lester D R, Herrera Hannah B, Lau Sean K
Southern California Permanente Medical Group, Department of Pathology, Woodland Hills Medical Center, 5601 De Soto Avenue, Woodland Hills, CA, 91365, USA.
Department of Pathology, Orange County-Anaheim Medical Center, Anaheim, CA, USA.
Head Neck Pathol. 2017 Dec;11(4):442-449. doi: 10.1007/s12105-017-0807-0. Epub 2017 Mar 14.
Thyroglossal duct cyst (TGDC) carcinomas are rarely encountered in the pediatric population. The clinical behavior of these tumors in the pediatric setting is unclear and management is not well defined. Two cases of pediatric thyroglossal duct cyst carcinoma were identified in a review of all thyroglossal duct cysts diagnosed over a ten year period. These two cases were analyzed along with 57 cases of thyroglossal duct cyst carcinoma affecting patients less than 21 years of age compiled from the English literature. Fifty-nine patients (36 females, 23 males) aged 6-20 years (mean 15.0 years) were identified. All presented with an anterior midline neck mass, which was typically mobile and non-tender. The average tumor size was 2.6 cm. Histologically, all tumors were papillary thyroid carcinomas arising in a background of a thyroglossal duct cyst. The tumors exhibited a papillary, follicular, or mixed architecture with classical papillary thyroid carcinoma nuclear features. Soft tissue extension was present in 16 cases. All patients were managed surgically with a Sistrunk procedure, with additional thyroidectomy performed in 29 patients, combined with a lymph node dissection (n = 15), or a Sistrunk and lymph node dissection (n = 5). All patients were stage I at presentation, with 11 showing lymph node metastases. Postoperative radioactive iodine was employed in 20 patients. A papillary carcinoma of the thyroid gland was reported in four of the patients who had concurrent/subsequent thyroidectomies. Recurrences were reported in four patients, with distant metastases in one patient, who died of disease (13 months). Follow up data was available for 45 patients, with an overall mean follow-up of 54.5 months. All patients were alive, with the exception of one who died with disease. TGDC carcinomas in pediatric patients is associated with a good overall prognosis, best managed by Sistrunk procedure alone, with selected lymph node dissection if clinically indicated.
甲状舌管囊肿（TGDC）癌在儿科人群中很少见。这些肿瘤在儿科环境中的临床行为尚不清楚，治疗方法也未明确界定。在对十年间诊断的所有甲状舌管囊肿进行回顾时，发现了两例儿科甲状舌管囊肿癌。这两例病例与从英文文献中收集的另外57例影响21岁以下患者的甲状舌管囊肿癌病例一起进行了分析。确定了59例年龄在6至20岁（平均15.0岁）的患者（36名女性，23名男性）。所有患者均表现为颈部前正中肿块，通常可活动且无压痛。平均肿瘤大小为2.6厘米。组织学上，所有肿瘤均为起源于甲状舌管囊肿背景的乳头状甲状腺癌。肿瘤表现为乳头状、滤泡状或混合结构，具有典型的乳头状甲状腺癌核特征。16例出现软组织浸润。所有患者均接受了Sistrunk手术治疗，29例患者额外进行了甲状腺切除术，其中15例联合淋巴结清扫，5例进行了Sistrunk手术和淋巴结清扫。所有患者就诊时均为I期，11例出现淋巴结转移。20例患者术后接受了放射性碘治疗。在同时进行/随后进行甲状腺切除术的4例患者中报告了甲状腺乳头状癌。4例患者出现复发，1例出现远处转移，该患者死于疾病（13个月）。45例患者有随访数据，总体平均随访时间为54.5个月。除1例死于疾病外，所有患者均存活。儿科患者的TGDC癌总体预后良好，仅通过Sistrunk手术治疗效果最佳，如有临床指征可进行选择性淋巴结清扫。
