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Turkish Journal of Cancer
2001, Volume 31, Number 3, Page(s) 121-124
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Primary non-Hodgkin's lymphoma of the oral tongue
GOPAL KRÝSHNA MAHESHWARI1, HARSHAD ACHARATLAL BABOO1, NILESH MANUBHAI SHAH2, MAHESH HIRJIBHAI PATEL3, RAKESH SHAH4
Departments of 1Radiation Oncology, 2Pathology, 3Surgical Oncology and 4Medical Oncology, The Gujarat Cancer and Research Institute, Ahmedabad-India
Keywords: non-hodgkin's lymphoma, oral tongue, extra-nodal lymphoma EXTRA-NODAL LYMPHOMA
Summary
Primary non-Hodgkin's lymphomas of the oral region are extremely rare. Only 12 cases of non-Hodgkin's lymphoma of the oral tongue have been reported in the literature (1-6). We describe a 30-year-old woman, who presented with a mass lesion primarily involving the oral tongue and diagnosed as diffuse large T -cell non-Hodgkin's lymphoma. The patient was successfully treated with wide excision of the lesion followed by chemotherapy and radiotherapy. She remained disease free 22- months after the diagnosis and 15-months following completion of treatment. This is the first case of non-Hodgkin's lymphoma of the oral tongue being reported from India.
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  • Case Presentation
    A 30-year-old-woman presented with a history of a slowly growing painless swelling on right side of the tongue for five-months. She had no other symptoms. Local examination revealed a 2x2 cm hard, nodular lesion involving the lateral margin of right half of the oral tongue (Figure 1). The mobility of the oral tongue was unaffected. Other parts of the oral cavity, oropharynx and neck as well as rest of the physical examination was normal. Her routine investigations including complete hemogram, urine analysis, chest X-ray and OPG (ortho-pantomogram) were normal. Biopsy of the lesion on histopathological examination demonstrated atypical large lymphoid cells infiltrating the skeletal muscles of the oral tongue (Figure 2). These cells had cleaved nuclei, moderate amount of eosinophilic cytoplasm, coarse chromatin and inconspicuous nucleoli. Moderate eosinophilic inflammatory infiltrate was also present in the background.

    A diagnosis of diffuse non-Hodgkin’s lymphoma was given. Immunohistochemical evaluation showed positive immunoreactivity for LCA (leukocyte common antigen), PAN-T (CD-3) and negative reaction with PAN-B (CD-20), cytokeratin (CK), AE1, Vimentin, S-100 and HMB-45. The patient was extensively investigated for other sites of involvement. Bone marrow aspiration, CSF examination, abdominal CT scan and ultrasonography were performed. No other sites in the body were found to be involved by the disease. Thus, a final diagnosis of the primary non-Hodgkin’s lymphoma, diffuse large cell type, T cell variety of the oral tongue was established and her disease was staged as I-E. She was HIV negative. Wide excision of the lesion was performed. All margins of the resection were free of the disease but base was involved. Lymphovascular invasion was absent. The patient was given six cycles of CHOP (Cyclophosphamide, vincristine, adriamycin and prednisone) chemotherapy followed by radiotherapy by parallel opposed lateral beams encompassing the orocervical region. She received 50 Gy in 25 fractions over a period of five weeks by 6 Mv photon beam on linear accelerator. There was moderate radiation reaction close to the completion of radiotherapy treatment. She currently remains disease free 22-months after diagnosis and 15-months following completion of treatment.

    Figure 1: Pre-treatment clinical photograph of the patient

    Figure 2: Micro-photographs of the lesion showing atypical large lymphoid cells infiltrating the skeletal muscle of the tongue. Moderate eosinophilic infiltrate is also noted in the background (A: H&E X100, B: H&E X400)

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  • Discussion
    Non-Hodgkin's lymphomas are a group of highly diverse malignancies and have great tendency to affect organs and tissues that do not ordinarily contain lymphoid cells. 20 to 30% of non-Hodgkin’s lymphoma arise from extranodal sites [7]. The head and neck is the second most common region for extranodal lymphoma after gastrointestinal tract [8,9]. Among various head and neck sites, Waldeyer's ring, which is an area encompassed by the nasopharynx, the tonsil and the base of the tongue is most often involved by malignant lymphoma [10,11]. The nose and paranasal sinuses, orbit(s), salivary glands are other sites in head and neck affected in descending order of frequency [12]. Involvement of various parts of the oral cavity is very uncommon [4,10]. The gingiva and the hard palate are the most often involved intraoral sites [13]. Involvement of buccal mucosa, oral tongue, floor of mouth and lip(s) has been reported quite infrequently. To date, only 12 cases of the non-Hodgkin’s lymphoma of the oral tongue has been mentioned in the literature [1-6]. This is the first case of non-Hodgkin's lymphoma of the oral tongue being reported from India.

    Little is known about the etiological factors for primary lymphoma of the oral region. Few cases of oral lymphomas have been reported in association with Acquired Immune Deficiency Syndrome (AIDS), and it may even be the first presentation of the disease in certain individuals [14-16]. It generally affects the elderly, especially over the 6th decade of life [17]. Our patient was 30-year-old woman. There are no characteristic clinical features of non-Hodgkin’s lymphoma of the oral region. The most common presenting symptoms are local swelling, pain or discomfort and ulcer. The oral non-Hodgkin’s lymphoma may mimic more commonly benign oral and dental pathologic conditions [10]. Thus, these lesions may be easily misdiagnosed. Awareness of this clinical entity in the oral region is important because lymphomas and more common malignant lesions such as carcinomas can not be differentiated clinically. Most of the head and neck non-Hodgkin's lymphomas including oral lesions are of B-cell origin and diffuse large cell type being the most common [18]. The present patient had diffuse T-cell variety, large cell type non-Hodgkin’s lymphoma.

    The paucity of cases makes the understanding of the biological behaviour and therapeutic options of lymphoma involving the oral region difficult. Like lymphomas at other head and neck sites, oral lesions also seem to be quite sensitive to both radiotherapy and chemotherapy. Our patient was treated with chemotherapy and radiotherapy following excision of the lesion as histopathology proved it to be a high grade lymphoma. She remained without evidence of disease 15-months after her treatment. The overall prognosis of non-Hodgkin's lymphoma is related to the stage of tumor and aggressiveness of the malignant cell type. In conclusion, though non-Hodgkin's lymphoma involving oral region is uncommon, it should always be considered in differential diagnosis of various benign and malignant lesions in this region, because the treatment and prognosis for these conditions are quite different. A proper clinical evaluation, histopathologic as well as immunohistochemical evaluation of biopsy specimen may aid in the diagnosis and thus, help in proper management.

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  • References

    1) Eisenbud L, Sciubba J, Mir R, et al. Oral presentations in non-Hodgkin's lymphoma: A review of 31 cases. Part I Data analysis. Oral Surg Oral Med Oral Path 1983;56:151-6.

    2) Touboul E, Ghenim C, Chantelar JV, et al. Lymphomes nonhodgkinies de la tete et du cou, stades I et II: A propos de 35 cas. Rev Stomatol Chir Maxillofac 1985;86:300-9.

    3) Haidar Z. A review of non-Hodgkin's lymphoma of the oral cavity 1950-1980. J Oral Med 1986;41:197-200.

    4) Wolvius EB, Van der Valk P, Vander Wal JE, et al. Primary extranodal non- Hodgkin's lymphoma of the oral cavity. An analysis of 34 cases. Eur J Cancer B Oral Oncol 1994;30B:121-5.

    5) Piattelli A, Di Alberti L, Artese L. Non-Hodgkin’s lymphoma of the tongue: a case report. Eur J Cancer B Oral Oncol 1996;32B:207-9.

    6) Dey P, Luthra UK, Sheikh ZA, et al. Fine needle aspiration cytology of primary non-Hodgkin’s lymphoma of the tongue. Acta Cytol 1999;43:422-4.

    7) Clearly KR, Batsakis JG. Sinonasal lymphomas. Ann Otol Rhinol Laryngol 1994;103:911-4.

    8) Freeman C, Berg JW, Cutler SJ. Occurrence and prognosis of extranodal lymphomas. Cancer 1972;29:252-60.

    9) Sutcliffe SB, Gospodarowicz MK. Localized extranodal lymphomas. In: Keating A, Armitage J, Burnett A, et al. editors. Hematological Oncology. Cambridge, England: Cambridge University Press, 1992;189-222.

    10) Wilson TG, Wright JM. Non-Hodgkin's lymphoma of the gingiva: review of the literature. Report of a case. J Periodontol 1986;57:155-7.

    11) Griffin TJ, Hurst PS, Swanson J. Non-Hodgkin's lymphoma: A case involving four third molar extraction sites. Oral Surg Oral Med Oral Pathol 1988;65:671-4.

    12) Kapadia SB, Barnes L, Deutsch M. Non-Hodgkin’s lymphoma of the nose and paranasal sinuses. A study of 17 cases. Head and Neck Surgery 1981;3:490-9.

    13) Scully C, Eveson JW, Witherow H, et al. Oral presentation of lymphoma: Case report of the T-cell lymphoma masquerading as oral Crohn's disease and review of the literature. Eur J Cancer B Oral Oncol 1993;29B:225-9.

    14) Silverman S, Migliori CA, Lozava-Nur F, et al. Oral findings in people with or at high risk for AIDS: A study of 375 homosexuals. J Am Dent Assoc 1986;112:187-92.

    15) Brahim JS, Katz RW, Roberts MW. Non-Hodgkin’s lymphoma of the hard palate mucosa and buccal gingiva associated with AIDS. J Oral Maxillofac Surg 1988;46:328-30.

    16) Rubin MM, Gatta CA, Cozzi GM. Non-Hodgkin’s lymphoma of the buccal gingiva as the initial manifestation of AIDS. J Oral Maxillofac Surg 1989;47:1311-3.

    17) Fukuda Y, Ishida T, Fujimoto M, et al. Malignant lymphoma of the oral cavity: Clinicopathologic analysis of 20 cases. J Oral Pathol 1987;16:8-12.

    18) Shima N, Kobashi Y, Tsutsui K, et al. Extranodal non-Hodgkin’s lymphoma of the head and neck. Cancer 1990;66:1190-7.

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