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 Table of Contents  
CASE REPORT
Year : 2018  |  Volume : 8  |  Issue : 2  |  Page : 116-119

Colonic phenotype of adenocarcinoma base of the tongue: An entity rarely reported and treated with definitive chemoradiation


1 Department of Radiation Oncology, Army Hospital Research and Referral, New Delhi, India
2 Department of Radiation Oncology, Command Hospital, Southern Command, Pune, Maharashtra, India
3 Department of Pathology and Molecular Science, Army Hospital Research and Referral, New Delhi, India

Date of Web Publication18-Dec-2018

Correspondence Address:
Abhishek Purkayastha
Department of Radiation Oncology, Command Hospital, Southern Command, Pune - 411 040, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijohs.ijohs_55_17

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  Abstract 


The predominant histological type of oropharyngeal cancer is squamous cell carcinoma and approximately 9 times more frequent when compared with other types. Adenocarcinoma common histopathology for digestive system is very rarely reported in this region. In literature, there are limited data about the clinical presentation, histopathology, and treatment modalities for the primary adenocarcinoma of the base of the tongue (BOT). To the best of our knowledge, five cases of adenocarcinoma of the BOT with colonic differentiation have been reported in the world literature till date, and this is the first case report from India. The challenge in these type of cases is to differentiate between the primary adenocarcinoma of BOT which is extremely rare, metastatic lesion to this site from any other site, or its origin from minor salivary glands which is a more common possibility because all these situations have different treatment implications.

Keywords: Adenocarcinoma, base of tongue, colonic type, oropharynx


How to cite this article:
Sharma N, Purkayastha A, Arora C, Sahai K. Colonic phenotype of adenocarcinoma base of the tongue: An entity rarely reported and treated with definitive chemoradiation. Int J Oral Health Sci 2018;8:116-9

How to cite this URL:
Sharma N, Purkayastha A, Arora C, Sahai K. Colonic phenotype of adenocarcinoma base of the tongue: An entity rarely reported and treated with definitive chemoradiation. Int J Oral Health Sci [serial online] 2018 [cited 2019 Mar 25];8:116-9. Available from: http://www.ijohsjournal.org/text.asp?2018/8/2/116/247813




  Introduction Top


Approximately 90% of oral cavity malignancies are squamous cell carcinoma (SCC). It is extremely rare to have a primary colonic type adenocarcinoma.[1] Immunohistochemistry (IHC) remains the gold standard for a definitive diagnosis. Given the rarity of the disease, there is no laid down the standard of treatment, and concurrent chemoradiation therapy (CCRT) has been tried and has proved beneficial.


  Case Report Top


A 71-year-old male presented with complaints of dysphagia and progressive painless swelling right side of the neck of 4-month duration. The patient's significant medical history included hypertension and a long history of smoking. Clinical evaluation revealed a firm 4.0 cm × 3.2 cm lesion limited to the left side base of the tongue (BOT) with indurated margins [Figure 1]. Neck examination revealed 2.5 cm × 3.0 cm right Level II lymph node and 2.0 cm × 1.5 cm left Level II and 1.5 cm × 2.0 cm left Level IV mobile lymph nodes.
Figure 1: Hopkins picture showing growth at the left side base of the tongue

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Evaluation with contrast-enhanced computed tomography (CT) scan showed a 4.6 cm × 3.5 cm soft-tissue mass lesion in the left BOT [Figure 2] with infiltration into genioglossus and partially into geniohyoid muscle. Direct laryngoscopy was done to rule out any second primary as a result of field cancerization in view of long-standing history of smoking in our patient. Extensive examination (colonoscopy and positron emission tomography/CT) failed to identify any gastrointestinal primary tumor.
Figure 2: Computed tomography scan head and neck showing a poorly defined heterogeneously enhancing soft tissue mass lesion epicentered in the left base of the tongue

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A biopsy was taken, and it revealed colonic-type adenocarcinoma [Figure 3]. Tumor in our patient was invasive well to moderately differentiated colonic-type adenocarcinoma. The colonic malignant epithelial cells forming tubular and glandular structures with intraglandular necrosis were dominantly observed in the tumor, with a significant mucinous component.
Figure 3: Biopsy from base of the tongue lesion showing well to moderately differentiated colonic-type adenocarcinoma (H and E, ×20)

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Immunohistochemically, the following primary antibodies were used by the ABC-method: CDX2, CK7, CK20, and beta-catenin. It revealed carcinoembryonic antigen (CEA) [Figure 4], human homeobox gene (CDX-2) highly positive [Figure 5], AE1/AE3 positive, and CK20+/CK7 status while synaptophysin, chromogranin, neuron-specific enolase, Melan-A, desmin, vimentin, CD 20 and CD99 were negative which suggested that the tumor did not originate from a minor salivary gland nor a metastasis from another primary. Therefore, it was considered as primary adenocarcinoma of BOT with colonic differentiation.
Figure 4: Immunohistochemistry showing carcinoembryonic antigen positivity being shown by increased intranuclear staining (×40)

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Figure 5: Immunohistochemistry showing CDX-2 positivity with brown nuclear staining (×40)

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The patient was staged as cT4aN2cM0 (Stage IV A) adenocarcinoma BOT and was treated with definitive CCRT because of the disease being surgically inoperable. The patient was treated with three-dimensional conformal radiotherapy to a dose of 70 Gy in 35 fractions along with concurrent chemotherapy cisplatin 35 mg/m2 weekly for a total of 6 cycles. At present, the patient is on three monthly follow-up for the last 2 years and is completely disease free.


  Discussion Top


We report a previously undocumented case in India [Table 1][2],[3],[4],[5] of primary colonic-type adenocarcinoma of the BOT. The challenge in these types of cases is to differentiate between primary adenocarcinoma of the BOT, metastatic lesion, or origin from minor salivary gland which is a more common possibility.[6] The oral cavity and oropharynx are very unusual sites of systemic metastases from primary cancers of other organs, which account for only about 1%–2% of oral malignant tumors as reported by Meyer and Shklar.[7] According to the literature, the most common primary sites which metastasize to oral or oropharyngeal sites are lung, kidney, and prostate for men while breast, reproductive tract, and kidney for women.[8] As per Shin et al.,[9] the histological features of most metastatic carcinomas were similar to those of primary tumors. Taken together, the lack of colonic primary tumors and the selective metastasis to the neck nodes formed the basis for the primary lingual origin of our case. Interestingly, the recent finding of adenocarcinoma in a foregut duplication cyst of the floor of the mouth of a 61-year-old male may suggest an alternative origin for this phenotype.[10] No evidence of a preexisting rudimentary cystic structure was found in our case. The histogenesis of this tumor is uncertain, but an origin from a transformed minor salivary duct epithelium at this location is a likely possibility.[11],[12],[13],[14],[15],[16] This is supported by the IHC findings in these tumors and by studies of primary intestinal-type adenocarcinoma of the sinonasal tract where evidence of respiratory epithelium transformation to an intestinal phenotype has been noted in the adjacent nonneoplastic epithelium.[17],[18],[19],[20],[21] The transdifferentiation of the respiratory to the intestinal type epithelium was associated with a reversal of the CK7/CK20 expression in the metaplastic epithelium which was maintained in the developing carcinoma.[17],[22] It is interesting, however, that similar primary phenotype in the lung manifested an immunoprofile consistent with primary adenocarcinoma and was negative for CK20 and CDX2.[20] These differences could be related to epigenetic differences between upper and lower respiratory epithelium.
Table 1: Previous reported cases in literature

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Bell et al. reported two such cases for the first time in the year 2009.[2] Both their cases manifested strong positive membranous/cytoplasmic staining for CK20 and beta-catenin and nuclear staining for CDX2. Additional intestinal markers done showed nuclear staining for hMLH1, hMSH2, hMSH6, and hPMS-2 mismatch repair proteins to be positive and DNA sequencing of codons 12, 13, and 61 of KRAS revealed no mutations. A case report by Solva et al.[3] also reported the similar type of IHC features for the colonic type of adenocarcinoma.

In a retrospective series from 1955 to 1985 of six cases by de Vries et al.,[23] all the adenocarcinomas had originated from minor salivary glands. In our case, the lymphatic pattern of spread was similar to BOT, and IHC was almost confirmatory. In contrast to our cases, all metastatic colon carcinomas to the tongue were preceded by colonic primary tumors in a series by Piattelli.[24] Solva et al.[3] managed their case with surgery both for primary and neck while Eskllzmir et al.[25] treated the patient both with surgery and external beam radiotherapy (EBRT). The patient could not be offered chemotherapy in view of the poor general condition. We offered radical CCRT to our patient as a definitive treatment on the lines of locally advanced SCC of BOT.

With no established treatment protocol for this type of clinical entity, we document its successful treatment by EBRT with concurrent chemotherapy though more prospective studies are required.

Acknowledgment

We thank the patient for allowing us to publish the case report and use the images taken during his stay in the hospital.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understand that name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Gillison ML. Current topics in the epidemiology of oral cavity and oropharyngeal cancers. Head Neck 2007;29:779-92.  Back to cited text no. 1
    
2.
Bell D, Kupferman ME, Williams MD, Rashid A, El-Naggar AK. Primary colonic-type adenocarcinoma of the base of the tongue: A previously unreported phenotype. Hum Pathol 2009;40:1798-802.  Back to cited text no. 2
    
3.
Slova D, Paniz Mondolfi A, Moisini I, Levi G, Urken M, Zevallos J, et al. Colonic-type adenocarcinoma of the base of the tongue: A case report of a rare neoplasm. Head Neck Pathol 2012;6:250-4.  Back to cited text no. 3
    
4.
Smith SM, Old M, Iwenofu OH. Primary lingual colonic-type adenocarcinoma: A Rare and emerging distinct entity! Head Neck Pathol 2017;11:234-9.  Back to cited text no. 4
    
5.
Rahimi S, Akaev I, Repanos C, Brennan PA, Dubois JD. Primary intestinal-type adenocarcinoma of tongue: A case report with immunohistochemical and molecular profiles and review of the literature. Head Neck Pathol 2017;11:186-91.  Back to cited text no. 5
    
6.
McDaniel AS, Burgin SJ, Bradford CR, McHugh JB. Pathology Quiz case 2. Diagnosis: Primary colonic-type adenocarcinoma of the tongue. JAMA Otolaryngol Head Neck Surg 2013;139:653-4.  Back to cited text no. 6
    
7.
Meyer I, Shklar G. Malignant tumors metastatic to mouth and jaws. Oral Surg Oral Med Oral Pathol 1965;20:350-62.  Back to cited text no. 7
    
8.
Hirshberg A, Shnaiderman-Shapiro A, Kaplan I, Berger R. Metastatic tumours to the oral cavity – Pathogenesis and analysis of 673 cases. Oral Oncol 2008;44:743-52.  Back to cited text no. 8
    
9.
Shin SJ, Roh JL, Choi SH, Nam SY, Kim SY, Kim SB, et al. Metastatic carcinomas to the ora l cavity and oropharynx. Korean J Pathol 2012;46:266-71.  Back to cited text no. 9
    
10.
Volchok J, Jaffer A, Cooper T, Al-Sabbagh A, Cavalli G. Adenocarcinoma arising in a lingual foregut duplication cyst. Arch Otolaryngol Head Neck Surg 2007;133:717-9.  Back to cited text no. 10
    
11.
Barnes L. Intestinal-type adenocarcinoma of the nasal cavity and paranasal sinuses. Am J Surg Pathol 1986;10:192-202.  Back to cited text no. 11
    
12.
Choi HR, Sturgis EM, Rashid A, DeMonte F, Luna MA, Batsakis JG, et al. Sinonasal adenocarcinoma: Evidence for histogenetic divergence of the enteric and nonenteric phenotypes. Hum Pathol 2003;34:1101-7.  Back to cited text no. 12
    
13.
Donhuijsen K, Hattenberger S, Schroeder HG. Nasal sinus carcinoma after wood dust exposure. Morphological spectrum of 100 cases. Pathologe 2004;25:14-20.  Back to cited text no. 13
    
14.
Franquemont DW, Fechner RE, Mills SE. Histologic classification of sinonasal intestinal-type adenocarcinoma. Am J Surg Pathol 1991;15:368-75.  Back to cited text no. 14
    
15.
Gnepp DR, Heffner DK. Mucosal origin of sinonasal tract adenomatous neoplasms. Mod Pathol 1989;2:365-71.  Back to cited text no. 15
    
16.
Hopkin N, McNicoll W, Dalley VM, Shaw HJ. Cancer of the paranasal sinuses and nasal cavities. Part I. Clinical features. J Laryngol Otol 1984;98:585-95.  Back to cited text no. 16
    
17.
Yom SS, Rashid A, Rosenthal DI, Elliott DD, Hanna EY, Weber RS, et al. Genetic analysis of sinonasal adenocarcinoma phenotypes: Distinct alterations of histogenetic significance. Mod Pathol 2005;18:315-9.  Back to cited text no. 17
    
18.
Bashir AA, Robinson RA, Benda JA, Smith RB. Sinonasal adenocarcinoma: Immunohistochemical marking and expression of oncoproteins. Head Neck 2003;25:763-71.  Back to cited text no. 18
    
19.
Ariza M, Llorente JL, Alvarez-Marcas C, Baragaño L, Salas A, Rodriguez Prado N, et al. Comparative genomic hybridization in primary sinonasal adenocarcinomas. Cancer 2004;100:335-41.  Back to cited text no. 19
    
20.
Kennedy MT, Jordan RC, Berean KW, Perez-Ordoñez B. Expression pattern of CK7, CK20, CDX-2, and villin in intestinal-type sinonasal adenocarcinoma. J Clin Pathol 2004;57:932-7.  Back to cited text no. 20
    
21.
Korinth D, Pacyna-Gengelbach M, Deutschmann N, Hattenberger S, Bockmühl U, Dietel M, et al. Chromosomal imbalances in wood dust-related adenocarcinomas of the inner nose and their associations with pathological parameters. J Pathol 2005;207:207-15.  Back to cited text no. 21
    
22.
Michal M, Skálová A, Simpson RH, Raslan WF, Curík R, Leivo I, et al. Cribriform adenocarcinoma of the tongue: A hitherto unrecognized type of adenocarcinoma characteristically occurring in the tongue. Histopathology 1999;35:495-501.  Back to cited text no. 22
    
23.
de Vries EJ, Johnson JT, Myers EN, Barnes EL Jr., Mandell-Brown M. Base of tongue salivary gland tumors. Head Neck Surg 1987;9:329-31.  Back to cited text no. 23
    
24.
Piattelli A. Lingual metastasis from a carcinoma of the colon. A case report and review of the literature. Acta Stomatol Belg 1990;87:257-64.  Back to cited text no. 24
    
25.
Eskllzmir G, Ozgur E, Tanyeri G, Ayhan S. Primary adenocarcinoma o the base of tongue: A rare case. J Med Updates 2014;4:81-5.  Back to cited text no. 25
    


    Figures

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