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 Table of Contents  
CASE REPORT
Year : 2016  |  Volume : 6  |  Issue : 2  |  Page : 92-95

Adenoid cystic carcinoma of mobile tongue


1 Department of Pathology, Sri Venkateswara Institute of Medical Sciences, Tirupati, Andhra Pradesh, India
2 Department of Surgical Oncology, Sri Venkateswara Institute of Medical Sciences, Tirupati, Andhra Pradesh, India
3 Department of Radiology, Sri Venkateswara Institute of Medical Sciences, Tirupati, Andhra Pradesh, India

Date of Web Publication13-Feb-2017

Correspondence Address:
Amitabh Jena
Department of Surgical Oncology, Sri Venkateswara Institute of Medical Sciences, Tirupati - 517 507, Andhra Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2231-6027.199983

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  Abstract 

Adenoid cystic carcinoma (ACC) is less frequently described in mobile tongue with approximate incidence of 3%. It takes its origin from the minor salivary glands and is malignant in nature. It is a slow-progressing tumor with high recurrence rate. Fine-needle aspiration cytology (FNAC) can play an important role in diagnosing palpable lesions of the lung. Hereby, we describe a rare case of ACC in a mobile tongue diagnosed by FNAC. The cytology smears revealed characteristic basaloid cells with hyperchromatic nuclei and there were numerous globules of basement membrane material. This 44-year-old lady underwent surgical excision with the final histopathological diagnosis of ACC.

Keywords: Adenoid cystic carcinoma, fine-needle aspiration cytology, minor salivary gland tumors, tongue


How to cite this article:
Patnayak R, Jena A, Inamdar MB, Reddy GV, Appasani S, Bodagala V. Adenoid cystic carcinoma of mobile tongue. Int J Oral Health Sci 2016;6:92-5

How to cite this URL:
Patnayak R, Jena A, Inamdar MB, Reddy GV, Appasani S, Bodagala V. Adenoid cystic carcinoma of mobile tongue. Int J Oral Health Sci [serial online] 2016 [cited 2017 Jul 21];6:92-5. Available from: http://www.ijohsjournal.org/text.asp?2016/6/2/92/199983


  Introduction Top


Adenoid cystic carcinoma (ACC) is a malignant neoplasm originating in both the minor and major salivary glands.[1] ACC is an uncommon lesion and accounts for 1%–2% of all head and neck malignancies and 10%–15% of all salivary gland malignancies.[2] ACC of mobile tongue is rare.[1],[2] The most common intraoral site for minor salivary gland tumors is the hard palate, followed by the base of the tongue.[1] Majority (96%) of the tumor in these locations are malignant.[1],[2] ACC represents about one-third of them.[1],[2] One of the least frequent sites of ACC is the mobile tongue, with a reported incidence of only approximately 3%.[1],[2]

Fine-needle aspiration cytology (FNAC) can play an important role in the diagnosis of oral lesions, particularly in those presenting as a mass or growth as tongue swellings are easily accessible to FNA.[3]

The present case is another case of ACC in a middle-aged female highlighting its cytological features in FNAC.


  Case Report Top


A 44-year-old lady presented with complaints of swelling on the undersurface of the tongue for 2 months. The onset of the swelling was insidious, and it gradually progressed to the present size of 2 cm × 2 cm. It was not associated with pain or dysphagia. She gave a history of palpitations. On examination of the oral cavity the soft to firm, nontender swelling was located on the ventral aspect of tongue, anterior 1/3rd in the midline, and more to the right side. The mucosa over the swelling was smooth and the margins were well defined [Figure 1]. There was no ankyloglossia and no other swelling in the oral cavity. There were no palpable lymph nodes in the neck. She was subjected to FNAC. The cytology smears were cellular and showed neoplastic cells with hyperchromatic nuclei showing acinar formation. There were many globules of basement membrane material [Figure 2] and [Figure 3]. The provisional diagnosis was ACC with differential diagnoses of polymorphous low-grade adenocarcinoma (PLGA) and pleomorphic adenoma (PA). Because of the presence of classical features such as characteristic basaloid cells with hyperchromatic nuclei and numerous globules of basement membrane material, it was reported as ACC.
Figure 1: Swelling ventral surface of tongue on the right side

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Figure 2: Cytosmear showing the presence of basaloid cells and globules of basement membrane material (May–Grunwald Giemsa stain, ×100)

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Figure 3: Cytosmear revealing basaloid cells with hyperchromatic nuclei and globules of basement membrane material (Papanicolouae stain, ×100)

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She underwent magnetic resonance imaging (MRI) of the tongue which was reported as the presence of a lesion of size 1.2 cm × 1.9 cm × 1.4 cm in the ventral aspect of anterior 1/3rd tongue [Figure 4]. The intrinsic muscles of tongue were involved on the right side. The right genioglossus muscle was also involved. Lesion was indenting lingual, septum on left side without infiltrating it. Her two-dimensional echocardiogram showed chronic rheumatic heart disease. Furthermore, she had severe aortic regurgitation, mild mitral regurgitation, and mild left ventricular dysfunction. The left ventricular ejection fraction was 46%. All other investigations were within normal limit. With antibiotic prophylaxis for rheumatic heart disease and under moderate cardiac risk, the patient underwent wide local excision and primary closure with bilateral supraomohyoid neck dissection. She recovered well and speech was almost normal.
Figure 4: (a and b) T1 and short tau inversion recovery coronal magnetic resonance images showing rounded T1 isointense and short tau inversion recovery hyperintense lesion in the anterior third of tongue involving intrinsic muscles

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The histopathology confirmed ACC with cribriform pattern. The neoplastic cells were small, with hyperchromatic nuclei and scant cytoplasm. Perineural infiltration was also present [Figure 5].
Figure 5: Histopathology of the tongue swelling showing cribriform pattern (Hematoxylin and Eosin, ×400)

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  Discussion Top


Although ACC is a common neoplasm of the salivary glands, it is rare in tongue and still rarer in mobile tongue.[1],[2] ACC predominantly occurs in females. Its peak incidence is in the fifth and sixth decades of life.[2] ACC is a slowly progressing tumor with high recurrence rate.[1],[2] ACC has strong association with neurotropism as evidenced by perineural invasion, reported in almost half of the cases.[1],[2] Local lymphnodal metastasis is uncommon. The frequent sites for distant metastasis are lung and bones.[1],[2]

Imaging modalities help to elicit the deep-seated lesions. MRI has higher accuracy in identifying the soft tissue lesion.[2]

Histologically, ACC can present three different variables: Glandular (cribriform), tubular, and solid.[4] The histological subtypes of lower grade malignancy (tubular and cribriform) have a better prognosis than those of high malignancy (solid).[1],[4]

The present case was reported as cribriform variant on histopathological examination with the presence of perineural infiltration. It showed numerous closely packed cystic spaces containing mucin lined by basaloid neoplastic cells (Swiss cheese pattern).

FNAC has been recommended as a valuable procedure for the initial evaluation of all intraoral lesions. It is fairly sensitive and specific procedure. Additional advantages are FNAC is simple, inexpensive, and comfortable to the patient. It helps provide a rapid diagnosis.[5]

Cytologically, the important differential diagnoses of ACC are PA and PLGA.

A well-defined cytoplasm, absent or few stripped nuclei and a bland finely granular nuclear chromatin indicates PA. On the other hand, scanty cytoplasm, a high Nuclear: Cytoplasm ratio, naked nuclei, nuclear molding, nuclear hyperchromasia, and coarseness favor ACC.[3] Problem may arise in those cases where smears are scant cellular with few hyaline globules. In our case, the presence of basaloid cells with hyperchromatic nuclei and the presence of numerous hyaline globules helped us in making the diagnosis of ACC on FNAC.

The presence of plasmacytoid appearance of individual tumor cells with abundant cytoplasm can differentiate PA from ACC.[3],[6] Ustündag et al. opined that the distinction of ACC from PA may not be easy always as myxoid acellular material and globules of basement membrane material may be noted in both the entities.[3],[7]

The cytodiagnosis and differentiation of PLGA from various other tumors are difficult as their cytological findings are not well characterized.[8] However, important clues toward a cytodiagnosis of PLGA are richly cellular smears containing a large number of cell clusters arranged in sheets or in a branching papillary pattern. The tumor cells exhibit moderate to abundant cytoplasm, round nuclei, fine chromatin, and inconspicuous nucleoli and minimal nuclear pleomorphism.[3] Surgery remains the cornerstone of treatment. Radiotherapy has a role for advanced T stages and as an adjuvant in the presence of positive microscopic margins.[1],[9] According to some authors, only advanced and nonresectable tumors may be treated with radiotherapy alone.[1],[10]


  Conclusion Top


ACC of the mobile tongue is an uncommon neoplasm. Early diagnosis is important in these are slowly growing tumors as they are prone for local invasion and possess high recurrence rate. FNA can play a role in diagnosing palpable lesions of tongue.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
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.  Back to cited text no. 1
    
2.
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.  Back to cited text no. 2
    
3.
Singh S, Garg N, Gupta S, Marwah N, Kalra R, Singh V, et al. Fine needle aspiration cytology in lesions of oral and maxillofacial region: Diagnostic pitfalls. J Cytol 2011;28:93-7.  Back to cited text no. 3
[PUBMED]  Medknow Journal  
4.
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.  Back to cited text no. 4
    
5.
Khan N, Afroz N, Haider A, Hassan MJ, Hashmi SH, Hasan SA. Role of fine needle aspiration, imprint and scrape cytology in the evaluation of intraoral lesions. J Cytol 2013;30:263-9.  Back to cited text no. 5
[PUBMED]  Medknow Journal  
6.
Singhal N, Khurana U, Handa U, Punia RP, Mohan H, Dass A, et al. Intraoral and oropharyngeal lesions: Role of fine needle aspiration cytology in the diagnosis. Indian J Otolaryngol Head Neck Surg 2015;67:381-7.  Back to cited text no. 6
    
7.
Ustündag E, Iseri M, Aydin O, Dal H, Almaç A, Paksoy N. Adenoid cystic carcinoma of the tongue. J Laryngol Otol 2000;114:477-80.  Back to cited text no. 7
    
8.
Sahai K, Kapila K, Dahiya S, Verma K. Fine needle aspiration cytology of minor salivary gland tumours of the palate. Cytopathology 2002;13:309-16.  Back to cited text no. 8
    
9.
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.  Back to cited text no. 9
    
10.
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.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]



 

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