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Year : 2019  |  Volume : 62  |  Issue : 3  |  Page : 454-456
Primary triple head and neck tumors: Laryngeal squamous cell carcinomas, Kaposi's sarcoma, and non-Hodgkin's lymphoma

1 Department of Otorhinolaryngology, Bagcilar Training and Research Hospital, Istanbul, Turkey
2 Department of Pathology, Bagcilar Training and Research Hospital, Istanbul, Turkey, India

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Date of Web Publication26-Jul-2019


Simultaneously triple head and neck malignancies are extremely rare. We report a case who had epithelial and mesenchymal malignant tumor with lymphoid malignancy in head and neck area. A patient who is 74 year old male patient presented to the otorhinolaryngology department with severe breathing difficulty due to laryngeal mass. The result of laryngeal biopsy was invasive SCCs, so patient underwent total larygectomy and bilateral level 2,3,4 neck disection operation. Primary 3 different type head and neck tumors were observed with histopathological examination. These were orderly invasive SCCs in larynx, B-cell Non-Hodgkin's lymphoma in tongue root and one lymph node of left neck dissection material, Kaposi's sarcoma in one lymph node of right neck dissection material. Although synchronous head and neck tumors occurs very rare with laryngeal carcinoma, the neck dissection materials should be researched for synchronous tumors.

Keywords: Head, Kaposi's sarcoma, lymphoma, neck neoplasms, squamous cell carcinoma

How to cite this article:
Yildirim M, Belli S, Ozsoy S, Taskin U. Primary triple head and neck tumors: Laryngeal squamous cell carcinomas, Kaposi's sarcoma, and non-Hodgkin's lymphoma. Indian J Pathol Microbiol 2019;62:454-6

How to cite this URL:
Yildirim M, Belli S, Ozsoy S, Taskin U. Primary triple head and neck tumors: Laryngeal squamous cell carcinomas, Kaposi's sarcoma, and non-Hodgkin's lymphoma. Indian J Pathol Microbiol [serial online] 2019 [cited 2020 Nov 28];62:454-6. Available from: https://www.ijpmonline.org/text.asp?2019/62/3/454/263488

   Introduction Top

Elective neck dissection is the current approach besides to laryngectomy in laryngeal carcinoma except T1-T2 glottic tumors. Histopathological examination of the neck dissection specimens in cancer surgery, and also other diseases, may occur except for metastatic lymphadenopathies.

These may include second primary tumors such as lymphoma, chronic infections such as tuberculosis, or inflammatory diseases such as sarcoidosis.

Synchronous tumors are tumors located in different regions simultaneously. Tumors should be physically distant from each other and the histological type should be different. Synchronous tumors of the same histological type require exclusion of metastasis. In literature, synchronous tumors such as Hodgkin's lymphoma and thyroid papillary carcinoma have been previously reported with larynx squamous cell carcinoma (SCC).[1],[2]

Kaposi's sarcoma and non-Hodgkin's lymphoma have not been reported simultaneously with larynx SCC.

Concurrent triple head and neck tumors are rare. We presented a case of larynx SCC with synchronous tumors such as Kaposi's sarcoma, which was located in the neck, and B-cell non-Hodgkin's lymphoma, which was located in the tongue root and neck.

   Case History Top

A 74 years old male patient was presented to the otorhinolaryngology department with a worsening condition of voice quality for the past 2 years and a breathing difficulty complaint for 3 months. Diffuse larynx edema, restriction of movement in the right vocal cord, left vocal cord fixation, and rima glottidis of approximately 2–3 mm size were observed in the endoscopic laryngeal examination. No mass or lymphadenopathy was found in the neck examination.

According to the contrast-enhanced neck MRI report, there is a soft tissue mass that begins from the left subglottic area and reaches the left vocal cord and left false vocal cord. It has a slight contrast enhancement in contrasted series. The lesion passes 1–2 mm deep into the anterior commissure and invades the paraglottic area of 2–3 mm. In addition, at the level of false vocal cord, there is an invasion of approximately 2–3 mm in paraglottic area measured at anterior–posterior length of 1.5 cm. Minimally increased contrast enhancement and mild heterogeneity are observed in the left aryepiglottic fold. There is another soft tissue lesion in the left vallecula with contrast enhancement of about 2.5 cm, extending around the left tongue root and left lingual tonsil (second tumor or submucosal infiltration is considered in differential diagnosis). Several reactive lymph nodes are observed of approximately 16 mm size in both proximal jugular chains.

We received informed consent from the patient to perform direct laryngoscopy and biopsy operation under general anesthesia. On the second day of hospitalization, multiple larynx biopsies were taken and tracheostomy was performed. As the result of the biopsy was invasive SCC, total laryngectomy and bilateral selective level 2, 3, and 4 neck dissection were performed on the seventh day of hospitalization.

Histopathological examination of the biopsy material showed that SCC was infiltrated into the left vocal cord, anterior commissure, left posterior commissure, and subglottic region [Figure 1]. Severe dysplasia and carcinoma in-situ areas in the right vocal cord and severe dysplasia in the left false cord were observed. Mild dysplasia and chronic inflammation were observed in the mucosa of the right aryepiglottic fold. Carcinoma in situ in left aryepiglottic fold mucosa and severe dysplasia in left piriform sinus mucosa were observed. Tumor surgical margin was negative. Perineural or lymphovascular invasion and thyroid cartilage invasion were not observed. SCC infiltration was not seen in the tongue root, and its distance to the tongue root was 2 cm. However, B-cell non-Hodgkin's lymphoma infiltration was observed. In the right neck dissection material, there were 29 reactive lymph nodes and 1 lymph node which contains Kaposi's sarcoma infiltration. Tumor cells showed diffuse H and E, CD34, and human herpesvirus 8 immunoreactivity [Figure 2]a, [Figure 2]b, [Figure 2]c. In the left neck dissection material, there were 24 reactive lymph nodes and 1 lymph node which contains non-Hodgkin's B-cell lymphoma infiltration. Atypical lymphoid cell proliferation with large vesicular nucleus was observed. Immunohistochemically, atypical lymphoid cells showed diffused CD20, CD10, and Bcl2 immunoreactivity, and CD5, CD3, and CD21 were not detected [Figure 3]a, [Figure 3]b, [Figure 3]c, [Figure 3]d. When the patient was investigated for lymphoma and Kaposi's sarcoma, there was not another area in the body. Adjuvant radiation was initiated to the patient. No recurrent mass or distant metastasis was detected in the patient's 2-year follow-up. We reported the case which showed the association of larynx SCC, Kaposi's sarcoma, and B-cell non-Hodgkin's lymphoma.
Figure 1: Squamous cell carcinoma, H and E ×100

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Figure 2: (a) Kaposi's sarcoma, H and E ×100; (b) Kaposi's sarcoma, CD34 ×100; and (c) Kaposi's sarcoma, HHV-8 ×100

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Figure 3: (a) Non-Hodgkin's lymphoma, H and E ×100; (b) non-Hodgkin's lymphoma, CD10 ×100; (c) non-Hodgkin's lymphoma, CD20 ×100; and (d) non-Hodgkin's lymphoma, Bcl2 ×100

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   Discussion Top

Larynx cancer is the most common head and neck cancer after skin tumors. Subglottic, supraglottic, and transglottic tumors often metastasizes to the cervical nodes, nevertheless this situation in glottic cancers is very rare. Although our case is of a transglottic tumor, cervical nodal metastasis is not seen.

The most common location of extranodal non-Hodgkin's lymphoma in the head and neck region is the Waldeyer's ring. Non-Hodgkin's lymphoma is seen in Waldeyer's ring such as eustachian tube, nasopharynx, oropharynx, soft palates, tonsils, and tongue.[3] 5–16% of all extranodal non-Hodgkin's lymphomas and 60–70% of head and neck extranodal non-Hodgkin's lymphomas occurs in Waldeyer's ring head and neck region.[4] B-cell non-Hodgkin's lymphoma has been detected in our patient on one lymph node in the neck and of approximately 2 × 2 cm size in left tongue root.

Kaposi's sarcoma is a malignant vascular tumor that is common in AIDS or other immunosuppressive conditions. It occurs by proliferation of endothelial or spindle cells. It is associated with HHV-8. These are of four types: classical, African-endemic, AIDS-associated, and immunosuppression and transplant-associated.[5] Kaposi's sarcoma occurs most commonly on hard palate and gingiva in the head and neck. It occurs also in buccal mucosa, lips, nasal cavity, submandibular duct, parotid gland, and infraparotid lymph glands.[6] There is no AIDS or immunosuppression in our case, and it is also located in the neck.

Warren and Gates described three criteria for multiple primer malignant tumors. Tumor histology should be malignant, each neoplasm should be physically separated (no submucosal and intraepithelial junctions), and metastasis should be excluded from the secondary tumor.[7]

Secondary primary tumors are common in the upper respiratory tract. Synchronous secondary primary tumors are seen among 9–14% of patients with head and neck cancer.

Joo et al. reported synchronous primary laryngeal carcinoma and Hodgkin's lymphoma.[2]

Triple head and neck tumors are very rare and two cases were encountered in the literature. Adams et al. reported a case of thyroid follicular cancer and thyroid papillary microcarcinoma associated with laryngeal carcinoma in a 69-year-old patient.[8] Ningombam et al. reported a case of thyroid papillary carcinoma and non-Hodgkin's lymphoma associated with larynx cancer in a 71-year-old patient.[1] Our case is the third one of triple primary head and neck tumors in the literature because of the laryngeal SCCs coexistence with tongue root, neck B-cell non-Hodgkin's lymphoma, and neck Kaposi sarcoma. In addition, there is no coexistence of laryngeal SCC, that is, epithelial malignant tumor, and Kaposi's sarcoma, that is, mesenchymal malignant tumor, in the literature.

   Conclusion Top

Although synchronous head and neck primary tumors are very rare with laryngeal carcinoma, the neck dissection materials should be researched for synchronous tumors. Synchronous tumors also affect the patient's morbidity and mortality; so, this reason makes them more crucial.

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Conflicts of interest

There are no conflicts of interest.

   References Top

Singh NJ, Tripathy N, Roy P, Manikantan K, Arun P. Simultaneous triple primary head and neck malignancies. Head Neck Pathol 2016;10:233-6.  Back to cited text no. 1
Joo YH, Jung CK, Sun DI, Kim MS. Synchronous laryngeal squamous cell carcinoma and Hodgkin lymphoma of the head and neck region. Auris Nasus Larynx 2009;36:501-4.  Back to cited text no. 2
Laskar S, Mohindra P, Gupta S, Shet T, Muckaden MA. Non-Hodgkin lymphoma of the Waldeyer's ring: Clinicopathologic and therapeutic issues. Leuk Lymphoma 2008;49:2263-71.  Back to cited text no. 3
Yong W, Zhang Y, Zheng W, Wei Y. Prognostic factors and therapeutic efficacy of combined radio-chemotherapy in Waldeyer's ring non-Hodgkin's lymphoma. Chin Med J 2000;113:148-50.  Back to cited text no. 4
Ramírez-Amador V, Anaya-Saavedra G, Martínez-Mata G. Kaposi's sarcoma of the head and neck: A review. Oral Oncol 2010;46:135-45.  Back to cited text no. 5
Mohanna S, Bravo F, Ferrufino JC, Sanchez J, Gotuzzo E. Classic Kaposi's sarcoma presenting in the oral cavity of two HIV-negative Quechua patients. Med Oral Patol Oral Cir Bucal 2007;12:365-8.  Back to cited text no. 6
Warren S, Gates O. Multiple primary malignant tumors: A survey of the literature and a statistical study. Am J Cancer 1932;16:1358-414.  Back to cited text no. 7
Adams M, Caffrey R. Triple primary cancers of the head and neck: Case report and literature review. J Laryngol Otol 2014;128:552-4.  Back to cited text no. 8

Correspondence Address:
Metin Yildirim
Bagcilar Training and Research Hospital, Otorhinolaryngology Department, Merkez Mh., Dr. Sadik Ahmet Caddesi, Istanbul 34100
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/IJPM.IJPM_555_18

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