Hamide SAYAR, İlhami YILDIRIM, Sevgi BAKARİŞ, Sülen SARIOĞLU, Hüseyin ÖZTARAKÇI

High-Grade Transformation of Adenoid Cystic Carcinoma Delineated with a Fibrous Rim: A Case Report

Background: High-grade transformation or dedifferentiation in carcinoma is progression of a low-grade malignant neoplasm to a high-grade carcinoma or poorly differentiated adenocarcinoma. This is rarely observed in adenoid cystic carcinoma of the salivary glands. Case Report: A 39 year-old woman presented with a painless mass at the left submandibulary region that had been growing slowly for 5 years. Submandibulary mass resection revealed a mass with peripheral adenoid cystic carcinoma and a central high-grade tumor delineated with a fibrous rim, raising the possibility of a hybrid or composite carcinoma, requiring differential diagnosis depending upon morphology and immunohistochemistry findings. The final histopathological diagnosis was high-grade transformation of adenoid cystic carcinoma. After surgical therapy, the patient was irradiated to the neck and submandibulary region. No sign of tumor recurrence has been evident for 36 months.Conclusion: This present case seems to be another rare case with high-grade transformation of adenoid cystic carcinoma and the fibrous rim may be a histopathological feature of such cases, which should be kept in mind.

PDF

___

1. Jaso J, Malhotra R. Adenoid cystic carcinoma. Arch Pathol Lab Med 2011;135:511-5.
2. Malhotra KP, Agrawal V, Pandey R. High grade transformation in adenoid cystic carcinoma of the parotid: report of a case with cytologic, histologic and immunohistochemical study. Head Neck Pathol 2009;3:310-4. [CrossRef]
3. Costa AF, Altemani A, Vékony H, Bloemena E, Fresno F, Suárez C, et al. Genetic profile of adenoid cystic carcinomas (ACC) with high-grade transformation versus solid type. Cell Oncol (Dordr) 2011;34:369-79. [CrossRef]
4. Seethala RR, Hunt JL, Baloch ZW, Livolsi VA, Leon Barnes E. Adenoid cystic carcinoma with high-grade transformation: a report of 11 cases and review of the literature. Am J Surg Pathol 2007;31:1683-94. [CrossRef]
5. Nagao T, Gaffey TA, Serizawa H, Sugano I, Ishida Y, Yamazaki K, et al. Dedifferentiated adenoid cystic carcinoma: a clinicopathologic study of 6 cases. Mod Pathol 2003;16:1265-72. [CrossRef]
6. Cheuk W, Chan JK, Ngan RK. Dedifferentiation in adenoid cystic carcinoma of salivary gland: an uncommon complication associated with an accelerated clinical course. Am J Surg Pathol 1999;23:465-72. [CrossRef]
7. Seethala RR, Cieply K, Barnes EL, Dacic S. Progressive genetic alterations of adenoid cystic carcinoma with high-grade transformation. Arch Pathol Lab Med 2011;135:123-30.
8. Evans HL. Liposarcoma: a study of 55 cases with a reassessment of its classification. Am J Surg Pathol 1979;3:507-23. [CrossRef]
9. Dahlin DC, Beabout JW. Dedifferentiation of low-grade chondrosarcoma. Cancer 1971;28:461-6. [CrossRef]
10. Chau Y, Hongyo T, Aozasa K, Chan JK. Dedifferentiation of adenoid cystic carcinoma: report of a case implicating p53 gene mutation. Hum Pathol 2001;32:1403-7. [CrossRef]
11. Ide F, Mishima K, Saito I. Small foci of high-grade carcinoma cells in adenoid cystic carcinoma represent an incipient phase of dedifferentiation. Histopathology 2003;43:604-6. [CrossRef]
12. Sato K, Ueda Y, Sakurai A, Ishikawa Y, Kaji S, Nojima T, et al. Adenoid cystic carcinoma of the maxillary sinus with gradual histologic transformation to high-grade adenocarcinoma: a comparative report with dedifferentiated carcinoma. Virchows Arch 2006;448:204-8. [CrossRef]
13. Kamio N, Tanaka Y, Mukai M, Ikeda E, Kuramochi S, Fujii M, et al. A hybrid carcinoma: adenoid cystic carcinoma and salivary duct carcinoma of the salivary gland. An immunohistochemical study. Virchows Arch 1997;430:495-500. [CrossRef]
14. Seethala RR. Histologic grading and prognostic biomarkers in salivary gland carcinomas. Adv Anat Pathol 2011;18:29-45. [CrossRef]