Indian Journal of Pathology and Microbiology
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Year : 2020  |  Volume : 63  |  Issue : 2  |  Page : 331-333
Squamous cell carcinoma larynx with concurrent Warthin's tumor of the submandibular gland: A rarely reported co-existence

1 Department of Pathology, AIIMS, Rishikesh, Uttarakhand, India
2 Department of Surgical Oncology, AIIMS, Rishikesh, Uttarakhand, India

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Date of Web Publication18-Apr-2020

How to cite this article:
Kumar U, Rajkumar B, Kumar A, Kishore S, Jat B. Squamous cell carcinoma larynx with concurrent Warthin's tumor of the submandibular gland: A rarely reported co-existence. Indian J Pathol Microbiol 2020;63:331-3

How to cite this URL:
Kumar U, Rajkumar B, Kumar A, Kishore S, Jat B. Squamous cell carcinoma larynx with concurrent Warthin's tumor of the submandibular gland: A rarely reported co-existence. Indian J Pathol Microbiol [serial online] 2020 [cited 2021 Jun 13];63:331-3. Available from: https://www.ijpmonline.org/text.asp?2020/63/2/331/282693

Dear Editor,

Squamous cell carcinoma of the larynx and hypopharynx is the second most common respiratory tract cancer, after lung cancer.[1] Surgery along with chemotherapy or radiotherapy is used for better prognosis in resectable cases. Surgery may be done with or without radical neck dissection. In cases where neck dissection is done a careful examination of the lymph node is warranted to rule out metastasis or any concurrent primary tumor. Any salivary gland submitted with lymph node should also be evaluated similarly. Warthin's tumor is most commonly located in the parotid gland and only a few cases have been reported in submandibular gland even in large studies.[2],[3],[4],[5] Presence of any lesion in this anatomical region may mimic clinically as metastatic lymphadenopathy.

A 52 years male patient presented with a complaint of change in voice for 8 months which was insidious in onset, progressive and then the patient was unable to produce voice. He also complained of difficulty in breathing and swallowing for 2 months which was insidious in origin and gradually progressive. History of bidi (tobacco) smoking two packs/day for 30 years was present. History of alcohol intake around 200 ml/day for the last 30 years was also present. On examination the patient was conscious and oriented to time, place, and person; vitals were stable. Bilateral carotid pulsation was present. Few subcentimetric lymph nodes were palpable on the bilateral level I, II, and III lymph nodes. Eastern Cooperative Oncology Group (ECOG) score was 1.

On laryngoscopic examination, an ulcer proliferative growth in supraglottis region involving right true and false vocal cords, aryepiglottic folds, arytenoid, and left arytenoid were noted. Laryngeal apparatus was mobile and laryngeal crepitations were absent.

The patient required urgent tracheostomy for dyspnea which was followed by Contrast-enhanced computed tomography (CECT) and punch biopsy of the lesion.

CECT of the neck showed a heterogeneously enhancing soft-tissue attenuation mass lesion measuring 6.7 × 3.7 cm involving the epiglottis, bilateral aryepiglottic folds and bilateral pyriform sinuses [Figure 1].
Figure 1: Contrast.enhanced computed tomography neck showing (a) mass lesion in larynx (b) mass in the larynx, submandibular gland, and level Ib lymph node

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The mass was infiltrating anteriorly into pre-epiglottic fat; inferiorly into bilateral false vocal cords, paraglottic fat, bilateral true vocal cords and bilateral arytenoids; superiorly into bilateral pharyngeal mucosal space; inferolaterally into right lamina of the thyroid cartilage and superficial lobe of the right submandibular gland. Posteriorly mass was abutting the paravertebral muscles. All other major structures including the tongue, base of the tongue, both parotids, and both lobes of the thyroid were normal.

Punch biopsy from growth supraglottis on histopathological examination was confirmatory for moderately differentiated squamous cell carcinoma.

A near-total laryngectomy and bilateral anterolateral neck dissection along with new glottis reconstruction were done. The surgical specimen was sent for histopathological examination.

On histopathology, we received a near-total laryngectomy specimen along with bilateral level I, II, III, IV lymph nodes and bilateral submandibular salivary glands. Grossly, 6 × 3.5 × 2.5 cm ulcer proliferative growth was seen involving supraglottis, glottis and subglottis. Histopathological examination showed moderately differentiated squamous cell carcinoma [Figure 2]. No metastatic deposit was identified in 30 lymph nodes examined. Right side salivary gland sent with level I lymph node showed double-layered epithelial cells on dense lymphocytic background diagnostic of Warthin's tumor [Figure 3]. Diagnosis of both squamous cell carcinoma and Warthin's tumor was histologically evident, and no ancillary testing was required.
Figure 2: Hematoxylin and eosin stain (a) Aryepiglottic fold showing dysplasia 200× (b and c) Glottis with tumor involvement showing keratin pearls and tumor infiltration 40× and 400× (d) Uninvolved arytenoid mucosa showing mild dysplasia and minor salivary glands 40× (e) Epiglottis showing hyperplastic and dysplastic epithelium 40× (f) Uninvolved thyroid gland 100×

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Figure 3: Warthin Tumor Hematoxylin and eosin stain (a) 40× (b) 100× (c) 400×

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A final diagnosis of moderately differentiated squamous cell carcinoma stage pT3N0Mx along with Warthin's tumor of the right submandibular gland was made.

Warthin's tumor of the submandibular gland is rare. In a case series of 2867 cases, of which 2513 were salivary gland tumor, only four cases were Warthin's tumor of the submandibular gland.[6] Coexisting Warthin's tumor with squamous cell carcinoma of the larynx has rarely been reported. In a large study of 78 cases of Warthin's tumor only two cases were associated with squamous cell carcinoma larynx.[7] Warthin's tumor of the submandibular gland may mimic tumor metastasis to the cervical lymph node.[8] Warthin's tumor has been classically associated with smoking. Clinical stage of tumor may be upgraded due to clinically apparent lymph node involvement. In this case, also lymph node status was considered to be 2c clinically and radiologically though none of the 30 lymph nodes examined histologically were involved by metastatic tumor.

Warthin's tumor of the submandibular gland is a rare finding and its presence with malignancy of the oral cavity or respiratory tract has been rarely reported. These findings need to be reported to ensure proper staging of tumor and to highlight the importance of proper sampling of the resected specimens. Moreover, extensive grossing of the specimen should be done if clinical lymph node stage is higher compared to the pathological stage to rule out the presence of any concurrent findings.

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Conflicts of interest

There are no conflicts of interest.

   References Top

El-Naggar AK, Chan JK, Rubin Grandis J, Takata T, Slootweg PJ. WHO Classification of Head and Neck Tumours. France, Lyon: International Agency for Research on Cancer; 2017. p. 159-202.  Back to cited text no. 1
Xu W, Lu H, Zhu Y, Ruan M, Zhang C, Yang W, et al. Warthin's tumour in oral and maxillofacial regions: An 18-year retrospective study of 1084 cases in an eastern-Chinese population. Int J Oral Maxillofac Surg 2018;47:913-7.  Back to cited text no. 2
Moeller K, Esser D, Boeger D, Buentzel J, Hoffmann K, Jecker P, et al. Parotidectomy and submandibulectomy for benign diseases in Thuringia, Germany: A population-based study on epidemiology and outcome. Eur Arch Oto-Rhino-Laryngology 2013;270:1149-55.  Back to cited text no. 3
Becerril-Ramírez PB, Bravo-Escobar GA, Prado-Calleros HM, Castillo-Ventura BB, Pombo-Nava A. Histología de tumores de glándulas submandibulares. Experiencia de 10 años. Acta Otorrinolaringológica Española 2011;62:432-5.  Back to cited text no. 4
Eveson JW, Cawson RA. Warthin's tumor (cystadenolymphoma) of salivary glands. A clinicopathologic investigation of 278 cases. Oral Surg Oral Med Oral Pathol 1986;61:256-62.  Back to cited text no. 5
Eneroth C-M. Salivary gland tumors in the parotid gland, submandibular gland, and the palate region. Cancer 1971;27:1415-8.  Back to cited text no. 6
Maiorano E, Lo Muzio L, Favia G, Piattelli A. Warthin's tumour: A study of 78 cases with emphasis on bilaterality, multifocality and association with other malignancies. Oral Oncol 2002;38:35-40.  Back to cited text no. 7
Chae SW, Sohn JH, Shin HS, Choi JJ, Kim YB. Unilateral, multicentric Warthin's tumor mimicking a tumor metastatic to a lymph node. Acta Cytol 2004;48:229-33.  Back to cited text no. 8

Correspondence Address:
Arvind Kumar
Department of Pathology, AIIMS, Rishikesh - 249 203, Uttarakhand
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/IJPM.IJPM_436_19

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  [Figure 1], [Figure 2], [Figure 3]


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