Mucoepidermoid carcinoma of the sublingual gland harboring a translocation of the MAML2 gene: A case report

Sato,Kiminobu; Akiba,Jun; Nakamura,Ken; Abe,Hideyuki; Kawahara,Akihiko; Aso,Takeichiro; Umeno,Hirohito; Harada,Hiroshi; Yano,Hirohisa

doi:10.3892/ol.2017.6550

Mucoepidermoid carcinoma of the sublingual gland harboring a translocation of the MAML2 gene: A case report

Authors:
- Kiminobu Sato
- Jun Akiba
- Ken Nakamura
- Hideyuki Abe
- Akihiko Kawahara
- Takeichiro Aso
- Hirohito Umeno
- Hiroshi Harada
- Hirohisa Yano
View Affiliations

Affiliations: Department of Otolaryngology‑Head and Neck Surgery, Kurume University School of Medicine, Kurume, Fukuoka 830‑0011, Japan, Department of Diagnostic Pathology, Kurume University Hospital, Kurume, Fukuoka 830‑0011, Japan, Department of Pathology, Kurume University School of Medicine, Kurume, Fukuoka 830‑0011, Japan, Department of Diagnostic Pathology, Seichokai Fuchu Hospital, Izumi, Osaka 594‑0076, Japan
Published online on: July 8, 2017 https://doi.org/10.3892/ol.2017.6550
Pages: 2970-2974
Copyright: © Sato et al. This is an open access article distributed under the terms of Creative Commons Attribution License.

Metrics: Total Views: 0 (Spandidos Publications: | PMC Statistics: )

Metrics: Total PDF Downloads: 0 (Spandidos Publications: | PMC Statistics: )

Cited By (CrossRef): 0 citations Loading Articles...

Abstract

Among tumors of the major salivary glands, tumors in the sublingual gland are rare. Although mucoepidermoid carcinoma (MEC) represents a histological type of salivary gland tumor, it is occasionally difficult to diagnose due to its histological variation. The present study reports a case of MEC harboring a mastermind‑like transcriptional coactivator 2 (MAML2) gene translocation in the sublingual gland. A 76‑year‑old Japanese woman with a mass in the left submandibular region was referred to Kurume University Hospital (Kurume, Japan). Computed tomography scans revealed that the tumor was predominantly located in the sublingual gland, and tumor resection was performed. Histologically, the tumor was composed of cells that exhibited low‑grade nuclear atypia and clear and/or granular eosinophilic cytoplasm, and that were proliferating in solid patterns. Periodic acid‑Schiff and alcian blue staining revealed a small number of mucinous cells in the tumor. Immunohistochemically, the tumor cells were positive for p40 and p63. Fluorescence in situ hybridization (FISH) analysis revealed a MAML2 gene split. The definitive pathological diagnosis was low‑grade MEC, as the case lacked any factors indicative of high‑grade malignancy. To the best of our knowledge, this is the first report of MEC in the sublingual gland with MAML2 gene translocation confirmed by FISH.

View Figures

View References

1	Eveson JW and Cawson RA: Salivary gland tumours. A review of 2410 cases with particular reference to histological types, site, age and sex distribution. J Pathol. 146:51–58. 1985. View Article : Google Scholar : PubMed/NCBI
2	Gary LE and Paul LA: Tumor of the salivary glandsAFIP Atlas of Tumor Pathology Series 4. AFIP ARP; Washington: pp. 173–196. 2008
3	Goode RK and El-Naggar AK: Mucoepidermoid carcinoma. In: World Health Organization Classification of TumorsPathology and Genetics of Head and Neck Tumors. Barnes L, Evenson JW, Reichart P and Sidransky D: IARC Press; Lyon: pp. 219–220. 2005
4	Stewart FW, Foote FW and Becker WF: Muco-epidermoid tumors of salivary glands. Am J Surg. 122:820–844. 1945.
5	John WE and Lester DRT: Malignant neoplasms of the salivary glandsHead and Neck Pathology. Lester DRT: 2nd. Elsevier Sauners; Philadelphia: pp. 270–277. 2013
6	Luna MA: Salivary mucoepidermoid carcinoma: Revisited. Adv Anat Pathol. 13:293–307. 2006. View Article : Google Scholar : PubMed/NCBI
7	Whatley WS, Thompson JW and Rao B: Salivary gland tumors in survivors of childhood cancer. Otolaryngol Head Neck Surg. 134:385–388. 2006. View Article : Google Scholar : PubMed/NCBI
8	Noda H, Okamura Y, Nakayama T, Miyabe S, Fujiyoshi Y, Hattori H, Shimozato K and Inagaki H: Clinicopathological significance of MAML2 gene split in mucoepidermoid carcinoma. Cancer Sci. 104:85–92. 2013. View Article : Google Scholar : PubMed/NCBI
9	Fehr A, Röser K, Heidorn K, Hallas C, Löning T and Bullerdiek J: A new type of MAMAL2 fusion in mucoepidermoid carcinoma. Genes Chromosomes Cancer. 47:203–206. 2008. View Article : Google Scholar : PubMed/NCBI
10	Goode RK, Alclair PL and Ellis GL: Mucoepidermoid carcinoma of the major salivary glands. Clinical and histopathologic analysis of 234 cases with evaluation of grading criteria. Cancer. 82:1217–1224. 1998. View Article : Google Scholar : PubMed/NCBI
11	Kumar AN, Nair PP, Thomas S, Raman PS and Bhambal A: Mucoepidemoid carcinoma of sublingual gland: A malignant neoplasm in an uncommon region. BMJ Case Rep May. 2011:bcr02201138642011.
12	Murase Y, Kawano S, Kiyoshima T, Goto Y, Matsubara R, Chikui T, Yoshiga D and Nakamura S: Case of mucoepidermoid carcinoma of the sublingual gland accompanied with extensive dystrophic calcification and intratumoral bone formation. Head Neck. 37:E161–E164. 2015. View Article : Google Scholar : PubMed/NCBI
13	Sumanth KN, Mainali A, Ongole R and Pai MR: Mucoepidermoid carcinoma: ‘A Mimicker’? J Nepal Dent Assoc. 10:31–34. 2009.
14	Furuse C, Sousa SO, Nunes FD, Magalhães MH and Araújo VC: Myoepithelial cell markers in salivary gland neoplasms. Int J Surg Pathol. 13:57–65. 2005. View Article : Google Scholar : PubMed/NCBI
15	Owosho AA, Aguilar CE and Seethala RR: Comparison of p63 and p40 (ΔNp63) as basal, squamoid, and myoepithelial markers in salivary gland tumors. Appl Immunohistochem Mol Morphol. 24:501–508. 2016. View Article : Google Scholar : PubMed/NCBI
16	Tajima S, Namiki I and Koda K: A clear cell variant of mucoepidermoid carcinoma harboring CRTC1-MAML2 fusion gene found in buccal mucosa: Report of a case showing a large clear cell component and lacking typical epidermoid cells and intermediate cells. Med Mol Morphol. 50:117–121. 2017. View Article : Google Scholar : PubMed/NCBI
17	Fujimaki M, Fukumura Y, Saito T, Mitani K, Uchida S, Yokoyama J, Yao T and Ikeda K: Oncocytic mucoepidermoid carcinoma of the parotid gland with CRTC1-MAML2 fusion transcript: Report of a case with review of literature. Hum Pathol. 42:2052–2055. 2011. View Article : Google Scholar : PubMed/NCBI
18	Wu L, Liu J, Gao P, Nakamura M, Cao Y, Shen H and Griffin JD: Transforming activity of MECT1-MAML2 fusion oncoprotein is mediated by constitutive CREB activation. EMBO J. 24:2391–2402. 2005. View Article : Google Scholar : PubMed/NCBI
19	Stenman G, Persson F and Andersson MK: Diagnostic and therapeutic implications of new molecular biomarkers in salivary gland cancers. Oral Oncol. 50:683–690. 2014. View Article : Google Scholar : PubMed/NCBI
20	Seethala RR and Chiosea SI: MAML2 status in mucoepidermoid carcinoma can no longer be considered a prognostic marker. Am J Surg Pathol. 40:1151–1153. 2016. View Article : Google Scholar : PubMed/NCBI
21	Spiro RH, Huvos AG, Berk R and Strong EW: Mucoepidermoid carcinoma of salivary gland origin. A clinicopathologic study of 367 cases. Am J Surg. 136:461–468. 1978. View Article : Google Scholar : PubMed/NCBI
22	Nance MA, Seethala RR, Wang Y, Chiosea SI, Myers EN, Johnson JT and Lai SY: Treatment and survival outcomes based on histologic grading in patients with head and neck mucoepidermoid carcinoma. Cancer. 113:2082–2089. 2008. View Article : Google Scholar : PubMed/NCBI
23	Elnayal A, Moran CA, Fox PS, Mawlawi O, Swisher SG and Marom EM: Primary salivary gland-type lung cancer: Imaging and clinical predictors of outcome. AJR Am J Roentgenol. 201:W57–W63. 2013. View Article : Google Scholar : PubMed/NCBI
24	Ishizumi T, Tateishi U, Watanabe S, Maeda T and Arai Y: F-18 FDG PET/CT imaging of low-grade mucoepidermoid carcinoma of the bronchus. Ann Nucl Med. 21:299–302. 2007. View Article : Google Scholar : PubMed/NCBI