Adenoid cystic carcinoma of the base of the tongue: Case report and literature review

Gozde Serindere; Gul Soylu Ozler; Sibel Hakverdi; Mehmet Serindere

doi:10.4103/ijohr.ijohr_21_17

CASE REPORT

Year : 2017 | Volume : 3 | Issue : 1 | Page : 36-39

Adenoid cystic carcinoma of the base of the tongue: Case report and literature review

Gozde Serindere¹, Gul Soylu Ozler², Sibel Hakverdi³, Mehmet Serindere⁴
¹ Department of Dentomaxillofacial Radiology, Faculty of Dentistry, Mustafa Kemal University, Ankara, Turkey
² Department of Otorhinolaryngology, Faculty of Medicine, Mustafa Kemal University, Hatay, Turkey
³ Department of Pathology, Faculty of Medicine, Mustafa Kemal University, Hatay, Turkey
⁴ Department of Radiology, Gulhane Education and Research Hospital, Ankara, Turkey

Date of Web Publication

17-Jul-2017

Correspondence Address:
Gozde Serindere
Department of Dentomaxillofacial Radiology, Faculty of Dentistry, Mustafa Kemal University, Hatay
Turkey

Source of Support: None, Conflict of Interest: None

Check

DOI: 10.4103/ijohr.ijohr_21_17

Abstract

Adenoid cystic carcinoma (ACC) is a rare malignant tumor arising from salivary glands. ACC of the head and neck has some features such as slow growth, high recurrence percentages, distant metastasis, and a tendency for perineural invasion. It comprises almost 2%–4% of head and neck area tumors. In minor salivary glands, it is generally observed in the palate. We report such a rare case that affected the base of the tongue in a 65-year-old-female patient and also carried out a brief literature review on the subject.

Keywords: Adenoid cystic carcinoma, base of tongue, head and neck cancer, minor salivary gland

How to cite this article:
Serindere G, Ozler GS, Hakverdi S, Serindere M. Adenoid cystic carcinoma of the base of the tongue: Case report and literature review. Indian J Oral Health Res 2017;3:36-9

How to cite this URL:
Serindere G, Ozler GS, Hakverdi S, Serindere M. Adenoid cystic carcinoma of the base of the tongue: Case report and literature review. Indian J Oral Health Res [serial online] 2017 [cited 2023 Dec 2];3:36-9. Available from: https://www.ijohr.org/text.asp?2017/3/1/36/210922

Introduction

Adenoid cystic carcinoma (ACC) was firstly determined by Robin, Lorian and Laboulbene in their articles published in 1853 and 1854, in these articles the cylindrical appearance of the tumor was mentioned. Billroth in 1859 defined the tumor as cylindroma and reported great recurrence rate of this tumor. Spies in 1930 termed as “adenoid cystic carcinoma” in his report of cutaneous and noncutaneous tumors of the basal cell type.^[1]

ACC is an uncommon epithelial tumor of the salivary glands. It accounts for about 5%–10% of all salivary gland neoplasms with 2%–4% of malignant occurrences of the head and neck region. About the 31% of lesions are seen in minor salivary glands, especially the palate, but besides that they may also be seen in the submandibular and parotid glands.^[2]

ACC has a widespread age distribution but is observed predominantly in women, between the fifth and sixth decades of life.^[3] Typical clinical findings contain local recurrence, slow growth, perineural invasion, and distant metastasis.^[4]

We present ACC of the base of tongue and a brief literature review on the report.

Case Report

A 65-year-old female patient had complained of a swelling in the median part of the tongue and pain in tongue for 10 months, which had started spontaneously and then showed an increase in size. She had difficulty with speech and swallowing. The patient reported an insignificant medical and family history except she had hypertension as systemic disease. Extraoral examination and palpation revealed cervical lymph node chain with normal size. On intraoral clinical examination, a firm sessile nodule of about 2 cm × 1 cm in diameter with a similar coloration to that of the buccal mucosa was observed [Figure 1]. There was pain in palpation. Based on patient's history and clinical examinations, associated with suspicious appearance of observed lesion with irregular form, malignant neoplasm was thought as early diagnosis.

Figure 1: Intraoral photograph showing a swelling on the base of the tongue

Click here to view

Magnetic resonance imaging (MRI) revealed an ill-defined, hypointense lesion on T1- and T2-weighted images [Figure 2]a and [Figure 1]b. The lesion enhanced markedly on postcontrast T1 with fat suppression [Figure 3]a and [Figure 3]b. Lesion was seen in left lateral and posterior aspect of the base of tongue extending up to lingual septum and crossing the midline. The lesion was involved the genioglossus and intrinsic muscles of the tongue base and infiltrated to posterior of left sublingual space. There was the indistinct border between the lesion and left lingual tonsil. There was oval shaped lymph node measured 10 mm × 5 mm in the cervical level 2b [Figure 4].

Figure 2: (a) T1-weighted and (b) T2-weighted axial scans showing an ill-defined, hypointense lesion

Click here to view

Figure 3: Postcontrast T1-weighted axial (a) and sagittal (b) images showing ill-defined heterogeneous signal intensity nodular mass lesion involving left lateral aspect of the tongue and extending up to lingual septum and crossing midline

Click here to view

Figure 4: Coronal scan showing the dimension of observed lymph node in the cervical level 2b

Click here to view

Due to hypointense lesion on T2, squamous cell carcinoma was eliminated, and we thought ACC and tongue of lymphoma as a differential diagnosis.

Soon after, the patient was referred to surgery, and the biopsy was performed, and specimen was sent for histopathologic study. [Figure 5]a shows the formation of tubular pattern that is characterized by eosinophilic content, infiltrating well-hyalinized background. There was perineural infiltration on serial sections [Figure 5]b. The immunohistochemical results revealed epithelial membrane antigen and Pan-CK in positive in tumor cells [Figure 6].

Figure 5: (a) Formation of tubular pattern is characterized by eosinophilic content, infiltrating well-hyalinized background (H and E, ×200). (b) Adenoid cystic carcinoma cells infiltrated along nerve sheath (H and E, ×200)

Click here to view

Figure 6: The tumor cells are positive with this stain (PAN-CK - IHC, ×200)

Click here to view

Based on the clinical, MRI, and histopathological findings, a final diagnosis was made as ACC of tongue base.

Discussion

Neoplasms of accessory salivary gland origin occur less commonly than major salivary glands. The tongue is a slightly uncommon region for salivary gland neoplasms.^[5]

ACC derives from both the minor and the major salivary glands. It is an uncommon lesion and constitutes about 1%–2% of all malignant neoplasms of the head and neck, and up to 10%–15% of all malignant salivary gland neoplasms. The most frequently seen intraoral region for minor salivary gland tumors is the hard palate, secondly the base of the tongue, up to 96% of all tumors are malignant, when the neoplasm is observed in tongue associatively and ACC comprises 30% of them.^[6] Similarly, in the study of Moran et al.,^[7] the hard palate is the most common region of this tumor, they reported that 9 of 38 cases of ACC in this area.

Eveson and Cawson ^[8] reported a discreet predominance of ACC cases in women (female:male 1.2:1) with ages varying from 24 to 78 years. Similarly, our patient was female and 65 years old.

In the clinical examination, ACC of the tongue is painless, slow submucosal growth which blocks the early diagnosis of ACC. Previous studies reported that the elapse time from the first clinical appearance to the entity of symptoms ranges from 2.5 to 7 years,^[9] with one case reported by Luna-Ortiz et al. that had a duration of 10 months.^[10] Similarly, in our case, the patient had pain and swelling within the duration of 10 months.

[Table 1] shows case series of ACC in tongue. In the study of Spiro et al.^[12] lesions involving accessory glands were observed in 171 patients of 242 salivary gland ACC cases while the palate was observed as the affected site in 64 patients (26%) and the tongue was reported as the second most affected area. Isacsson and Shear ^[13] reported neoplasm occurrences in the palate, floor of the mouth, tongue, and gingiva, respectively. Khan et al.^[16] reported that ACC originates from major salivary glands in 26 of 68 cases and from minor salivary glands in rest of them, and 5 ACC cases in tongue of 68 cases.

Table 1: Case series of adenoid cystic carcinoma in tong

Click here to view

MRI has higher accuracy in describing the soft tissue lesion.^[18] For this reason, in our case, MRI was used as an imaging modality.

Histologically, ACC has three different types as cribriform, tubular, and solid. Worst prognosis was based on the presence of increased mitotic figures. It also has a strong neurotrophism including the nerves neighboring on the lesion.^[19] Soares et al.^[19] and Batsakis et al.^[20] reported that survival rate was less in the solid pattern than the cribriform and tubular patterns in their studies about the relationship between the histological pattern and the prognosis.

Surgical excision of the tumor with adjuvant radiotherapy in patients with advanced T stage and/or positive surgical margins is the treatment choice.^[21] The deficiency of survival advantage for patients treated with combination surgery and radiotherapy is presumed to be because of the high rate of distant metastases and the relatively high possibility of long-term survivability after salvage therapy for patients who experienced locoregional recurrence.^[22] Nascimento et al.^[15] reported that local recurrence was identified in 17 (37%) of the 46 patients.

Patients who have ACC in the tongue complain insufficient function. Thereby improvement the speech and swallowing of the patient must be the purpose of the treatment. As a result, the quality of life for the patient will increase.

Conclusion

ACC of the tongue is a very rare neoplasm and early diagnosis and suitable treatment are important and remarkable factors. Tongue lesions involving ACC is difficult for diagnosis and surgical treatment because of their anatomical location and the adjacency with blood vessels and nerves. In the literature, tongue has the low percentage as the sites of ACC to make this case a rare one. The histopathological study and the presence or absence of distant metastases are important to find final diagnose and describe the survival of the patient.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

References

1.	Shankar NV, Prakash SM, Sumalatha MN, Shankar A. Adenoid cystic carcinoma of the tongue. Int J Acad Res 2011;3:580-3.
2.	Triantafillidou K, Dimitrakopoulos J, Iordanidis F, Koufogiannis D. Management of adenoid cystic carcinoma of minor salivary glands. J Oral Maxillofac Surg 2006;64:1114-20. [PUBMED]
3.	Waldron CA, el-Mofty SK, Gnepp DR. Tumors of the intraoral minor salivary glands: A demographic and histologic study of 426 cases. Oral Surg Oral Med Oral Pathol 1988;66:323-33. [PUBMED]
4.	Huang M, Ma D, Sun K, Yu G, Guo C, Gao F. Factors influencing survival rate in adenoid cystic carcinoma of the salivary glands. Int J Oral Maxillofac Surg 1997;26:435-9. [PUBMED]
5.	Goldblatt LI, Ellis GL. Salivary gland tumors of the tongue. Analysis of 55 new cases and review of the literature. Cancer 1987;60:74-81. [PUBMED]
6.	Luna-Ortiz K, Carmona-Luna T, Cano-Valdez AM, Mosqueda-Taylor A, Herrera-Gómez A, Villavicencio-Valencia VV. Adenoid cystic carcinoma of the tongue clinicopathologic study and survival analysis. Head Neck Oncol 2009;1:15.
7.	Moran JJ, Becker SM, Brady LW, Rambo VB. Adenoid cystic carcinoma. A clinicopathological study. Cancer 1961;14:1235-50. [PUBMED]
8.	Eveson JW, Cawson RA. Tumours of the minor (oropharyngeal) salivary glands: A demographic study of 336 cases. J Oral Pathol 1985;14:500-9. [PUBMED]
9.	Carrasco Ortiz D, Aldape Barrios B. Adenoid cystic carcinoma of the dorsum of the tongue: Presentation of a case. Med Oral Patol Oral Cir Bucal 2006;11:E417-20. [PUBMED]
10.	Luna Ortiz K, Carmona Luna T, Herrera Gómez A, Cano Valdez AM. Macroglossia caused by adenoid cystic carcinoma. Case report. Med Oral Patol Oral Cir Bucal 2008;13:E395-7.
11.	Leafstedt SW, Gaeta JF, Sako K, Marchetta FC, Shedd DP. Adenoid cystic carcinoma of major and minor salivary glands. Am J Surg 1971;122:756-62. [PUBMED]
12.	Spiro RH, Huvos AG, Strong EW. Adenoid cystic carcinoma of salivary origin. A clinicopathologic study of 242 cases. Am J Surg 1974;128:512-20. [PUBMED]
13.	Isacsson G, Shear M. Intraoral salivary gland tumors: A retrospective study of 201 cases. J Oral Pathol 1983;12:57-62. [PUBMED]
14.	Dal Maso M, Lippi L. Adenoid cystic carcinoma of the head and neck: A clinical study of 37 cases. Laryngoscope 1985;95:177-81. [PUBMED]
15.	Nascimento AG, Amaral AL, Prado LA, Kligerman J, Silveira TR. Adenoid cystic carcinoma of salivary glands. A study of 61 cases with clinicopathologic correlation. Cancer 1986;57:312-9. [PUBMED]
16.	Khan AJ, DiGiovanna MP, Ross DA, Sasaki CT, Carter D, Son YH, et al. Adenoid cystic carcinoma: A retrospective clinical review. Int J Cancer 2001;96:149-58. [PUBMED]
17.	da Cruz Perez DE, de Abreu Alves F, Nobuko Nishimoto I, de Almeida OP, Kowalski LP. Prognostic factors in head and neck adenoid cystic carcinoma. Oral Oncol 2006;42:139-46. [PUBMED]
18.	Baskaran P, Mithra R, Sathyakumar M, Misra S. Adenoid cystic carcinoma of the mobile tongue: A rare case. Dent Res J (Isfahan) 2012;9 Suppl 1:S115-8.
19.	Soares EC, Carreiro Filho FP, Costa FW, Vieira AC, Alves AP. Adenoid cystic carcinoma of the tongue: Case report and literature review. Med Oral Patol Oral Cir Bucal 2008;13:E475-8. [PUBMED]
20.	Batsakis JG, Luna MA, el-Naggar A. Histopathologic grading of salivary gland neoplasms: III. Adenoid cystic carcinomas. Ann Otol Rhinol Laryngol 1990;99:1007-9. [PUBMED]
21.	Silverman DA, Carlson TP, Khuntia D, Bergstrom RT, Saxton J, Esclamado RM. Role for postoperative radiation therapy in adenoid cystic carcinoma of the head and neck. Laryngoscope 2004;114:1194-9. [PUBMED]
22.	Mendenhall WM, Morris CG, Amdur RJ, Werning JW, Hinerman RW, Villaret DB. Radiotherapy alone or combined with surgery for adenoid cystic carcinoma of the head and neck. Head Neck 2004;26:154-62. [PUBMED]