Rutigliano Daniel N, Meyers Paul, Ghossein Ronald A, Carlson Diane L, Kayton Mark L, Kraus Dennis, La Quaglia Michael P
Department of Surgery, Division of Pediatric Surgery, Memorial Sloan-Kettering Cancer Center, New York, NY 10021, USA.
J Pediatr Surg. 2007 Jul;42(7):E9-13. doi: 10.1016/j.jpedsurg.2007.04.028.
Children diagnosed with osteosarcoma (OS) and Ewing sarcoma (ES) have greatly benefited from the addition of alkylator therapy. However, with greater numbers of long-term survivors, the rising incidence of secondary malignant neoplasms (SMNs) is concerning. Herein we report on 2 patients with sarcoma who developed a case of secondary mucoepidermoid carcinoma after chemotherapy treatment without associated radiation therapy. To our knowledge, this is the first series of mucoepidermoid carcinomas arising in pediatric patients treated for sarcoma without radiotherapy.
Long-term survivors of OS and ES currently undergoing routine follow-up care were reviewed and noted for the development of a new secondary malignancy. Details of their initial evaluation, previous therapies, resection techniques, pathologic findings, and follow-up compose this report.
Two patients, a 17-year-old adolescent boy with OS and 16-year-old adolescent girl with ES, with secondary mucoepidermoid carcinoma of the parotid gland were identified. Both patients underwent primary resection and chemotherapy including alkylating agents, but neither received radiation. The mucoepidermoid carcinomas developed 27 months and 132 months after completion of therapy, respectively, and were noted on routine yearly follow-up. Fine-needle aspiration was nondiagnostic on each, and parotidectomy with preservation of the facial nerve was performed. Pathology revealed low-grade mucoepidermoid carcinoma with tumor extending to the deep margins for both lesions, and radiotherapy to the parotid bed was administered. There were no surgical complications. One patient is alive, without evidence of recurrent mucoepidermoid carcinoma after 4 years; the other recently completed radiotherapy and is disease-free after 12 months.
Primary mucoepidermoid carcinoma of the parotid gland accounts for less than 10% of all head and neck tumors in childhood. Previous series of secondary mucoepidermoid carcinoma have demonstrated an increased risk in patients with leukemia/lymphoma. This is the first reported series of parotid mucoepidermoid carcinomas occurring after sarcoma treatment without radiotherapy. A common link between the 2 patients may be the use of alkylating therapy.
诊断为骨肉瘤（OS）和尤因肉瘤（ES）的儿童因加入烷化剂治疗而受益匪浅。然而，随着长期存活者数量的增加，继发性恶性肿瘤（SMN）发病率的上升令人担忧。在此，我们报告2例肉瘤患者，他们在化疗后发生了继发性黏液表皮样癌，且未接受相关放疗。据我们所知，这是首例在未接受放疗的肉瘤儿科患者中发生的黏液表皮样癌系列病例。
对目前正在接受常规随访的骨肉瘤和尤因肉瘤长期存活者进行回顾，并记录新发性继发性恶性肿瘤的发生情况。本报告包括他们的初始评估、既往治疗、切除技术、病理结果及随访的详细信息。
确定了2例患者，1例为17岁患骨肉瘤的青少年男性，1例为16岁患尤因肉瘤的青少年女性，均患有腮腺继发性黏液表皮样癌。两名患者均接受了包括烷化剂在内的原发肿瘤切除及化疗，但均未接受放疗。黏液表皮样癌分别在治疗结束后27个月和132个月发生，在每年的常规随访中被发现。细针穿刺活检对二者均未明确诊断，遂行保留面神经的腮腺切除术。病理显示为低级别黏液表皮样癌，两个病灶的肿瘤均延伸至深部边缘，并对腮腺床进行了放疗。无手术并发症。1例患者存活，4年后无复发性黏液表皮样癌证据；另1例最近完成放疗，12个月后无疾病复发。
腮腺原发性黏液表皮样癌在儿童所有头颈部肿瘤中占比不到10%。既往关于继发性黏液表皮样癌的系列研究表明白血病/淋巴瘤患者风险增加。这是首例报道的肉瘤治疗后未行放疗而发生的腮腺黏液表皮样癌系列病例。这2例患者的一个共同联系可能是使用了烷化剂治疗。
