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CORRESPONDENCE
Year : 2014  |  Volume : 10  |  Issue : 2  |  Page : 407-409

MALT lymphoma at the base of tongue of a 29-year-old woman treated with radiation therapy alone


1 Department of Radiation Oncology, Seoul St. Mary's Hospital, College of Medicine, The Catholic University Medical College, Seoul, Korea
2 Department of ENT Head and Neck Surgery, Seoul St. Mary's Hospital, College of Medicine, The Catholic University Medical College, Seoul, Korea
3 Department of Internal Medicine, Seoul St. Mary's Hospital, College of Medicine, The Catholic University Medical College, Seoul, Korea
4 Hospital Pathology, Seoul St. Mary's Hospital, College of Medicine, The Catholic University Medical College, Seoul, Korea

Date of Web Publication14-Jul-2014

Correspondence Address:
Yeon Sil Kim
Department of Radiation Oncology, Seoul St. Mary's Hospital, College of Medicine, The Catholic University Medical College, 505 Banpo-dong, Seocho-gu, Seoul - 137 701
Korea
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0973-1482.136673

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 > Abstract 

Mucosa-associated lymphoid tissue (MALT) lymphoma is frequently reported in the gastrointestinal tract (GIT), but the incidence is low in the upper aerodigestive tract. In particular, MALT lymphoma of the tongue is very rare. Only four cases have been reported in the English literature to date. We report a case of 29-year-old woman who had a past history of peripheral T cell lymphoma of the head and neck and a new mass at the right base of tongue 3 years later. An incisional biopsy of the base of tongue revealed a new pathology, one that of extranodal marginal zone B cell lymphoma (MALT lymphoma). After staging work-up, she was diagnosed to be at the Ann Arbor stage IE. She was treated with 30.6 Gy of radiation therapy alone and there was no recurrence after 3 years follow-up.

 > Abstract in Chinese 

一个29岁的女人舌根部MALT淋巴瘤,行单纯放疗
摘要
黏膜相关淋巴组织(MALT)淋巴瘤是经常在胃肠道(GIT)的报道,但发生在上呼吸消化道低。特别是,舌头的MALT淋巴瘤是非常罕见的。英国文学资料只有四例病例报道。我们报告一例29岁的妇女,头部和颈部有外周T细胞淋巴瘤的过去史,3年之后在舌根疗出现新的肿块。舌根切取活检显示新的病理,一个结外边缘区B细胞淋巴瘤(MALT淋巴瘤)。分期后,她被确诊在Ann Arbor分期IE。予单纯放疗,剂量30.6gy,治疗后随访3年,无复发。
关键词:舌根,黏膜相关淋巴组织淋巴瘤,放疗


Keywords: Base of tongue, mucosa-associated lymphoid tissue lymphoma, radiation therapy


How to cite this article:
Song JH, Sun DI, Hong YS, Park GS, Kim YS. MALT lymphoma at the base of tongue of a 29-year-old woman treated with radiation therapy alone. J Can Res Ther 2014;10:407-9

How to cite this URL:
Song JH, Sun DI, Hong YS, Park GS, Kim YS. MALT lymphoma at the base of tongue of a 29-year-old woman treated with radiation therapy alone. J Can Res Ther [serial online] 2014 [cited 2019 Nov 20];10:407-9. Available from: http://www.cancerjournal.net/text.asp?2014/10/2/407/136673


 > Introduction Top


Non-Hodgkin's lymphoma (NHL) is a malignant tumor arising from the lymph nodes and/or other lymphoid tissues. The primary site is usually lymph nodes in three-fourth of patients, but in others it can be extranodal lymphoid tissues. Lymphoid tissue can be also formed in the organs that have mucosa; such as digestive tracts, bronchial tracts, and salivary glands. This formation is called mucosa-associated lymphoid tissue (MALT). [1] MALT lymphoma is an extranodal malignant tumor arising from this tissue. Since the time it was first reported in 1983 by Isaacson and Wright, this category of disease was not included in the Working Formulation for Clinical Usage, until it was reclassified as a part of 'the marginal zone B cell lymphoma' in the REAL classification in 1994. [2],[3] MALT lymphoma is frequently reported in the gastrointestinal tract (GIT), but is rare in the upper aerodigestive tract, with only four cases of MALT lymphoma of tongue reported to our knowledge. [4],[5],[6],[7]

We report a case of 29-year-old woman with a past history of peripheral T cell lymphoma, who developed a MALT lymphoma in the base of tongue 3 years later and was successfully treated with radiation therapy.


 > Case report Top


A 29-year-old woman with odynophagia visited our hospital. She had been previously diagnosed with peripheral T cell lymphoma (PTCL) 3 years ago and had been successfully treated with chemotherapy. She had no other special past, social, and family history.

At the time when she was diagnosed with PTCL, her chief complaint had been palpable masses on both sides of the neck. Physical examination revealed a bulging mass on the left side of the oropharyngeal wall and, two hard and fixed masses of 3 and 7 cm in length were palpable on both sides neck. PTCL was diagnosed in the excised mass from the right neck. The immunohistochemistry (IHC) results showed CD45RO (+), CD20 (−), CD3 (−), CD15 (−), CD30 (−), and molecular cytogenetics revealed positive results indicating rearrangement in the T cell receptor (TCR) gene [Figure 1]. The neck computed tomography (CT) and positron emission tomography-CT (PET-CT) results showed that the mass mainly involved areas from the left nasopharyngeal to oropharyngeal lateral wall. Multiple enlarged lymph nodes were observed along both internal jugular neck chains. There were no other specific findings in the staging work up, so she was diagnosed as PTCL, of the Ann Arbor stage II. She was treated chemotherapy consisting of six cycles of rituximab-cyclophosphamide, doxorubicin, vincristine, and prednisone (R-CHOP). Complete remission was achieved after chemotherapy and regular follow-up studies showed no evidences of recurrence.
Figure 1: The histopathology of peripheral T cell lymphoma. The specimen was from the neck lymph node. (a) Diffuse infiltrates of large atypical lymphoid cells with pleomorphic nuclei. The lymphoma cells are (b) positive for CD45RO, a T cell marker, (c) negative for CD20, a B cell marker and (d) negative for CD3, a primitive T cell marker

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Three years later, she visited our hospital again with odynophagia that had been progressed for 1 month. Physical examination and CT of the neck showed a new mass arising from the right base of tongue which also involved the right oropharyngeal wall [Figure 2]. An incisional biopsy showed a different pathology from the previous disease but one that of an extranodal marginal zone B cell lymphoma (MALT lymphoma). The IHC results were CD45RO (−), CD20 (+), CD79a (+), CD3 (−) and Bcl2 (+) [Figure 3], which corresponded to MALT lymphoma.
Figure 2: Fiberscopy and neck CT findings of mucosa-associated lymohoid tissue lymphoma. (a) A prominent hypodense lobulate lesion involves the right base of tongue and the oropharyngeal wall. (b) No abnormal lymph nodes were seen

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Figure 3: The histopathology of MALT lymphoma. The specimen was from the tongue base biopsy. (a) Diffuse infiltrates of abnormal lymphoid cells in the epithelium, mainly consisting of centrocyte-like cells. Immunostaining are (b) positive for CD20, a B cell marker, (c) negative for cytokeratin, an epithelial cell marker (only the epithelial cells show positive results) and (d) negative for CD3, a T cell marker

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Endoscopy; bone marrow biopsy; and CT of the neck, chest and abdomen were performed again for new staging. There was no evidence of regional or distant metastasis, and she was diagnosed as having extranodal marginal zone B cell lymphoma on the base of tongue of the Ann Arbor stage IE. The oncologist consulted our department for radical radiation therapy.

In simulation for radiation therapy, the neck was immobilized with an aquaplast mask and CT scan with contrast enhancement was performed. The main tongue mass was contoured as GTV (gross tumor volume) and the neck lymphatic chains from level I to level V was contoured as clinical target volume (CTV). The planning target volume was defined with a CTV plus 1.0-1.5 cm margin. The treatment plan was designed with bilateral fields in the upper neck (above the hyoid bone) and an anterior port with midline shield in the lower neck [Figure 4]. Six MV photons were used in the treatment. She was treated daily with from 180 cGy up to 3060 cGy of radiation, five times a week. Chemotherapy was not given.
Figure 4: A simulation film of radiation treatment field and isodose line display. (a) Radiation fields consisted of two bilateral fields in the upper neck and an anterior port with a midline shield in the lower neck, (b) An isodose line is displayed in entire neck region

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During the radiation therapy, she complained only of Grade I xerostomia. Other acute side effects, such as mucositis or decreased performance, did not occur. Not only the symptoms of odynophagia, but also xerostomia disappeared after 2-months' follow-up. Physical examination and imaging studies taken after 3 months of the completion of radiation therapy showed that the mass had regressed completely. Complete remission state was maintained in 38 months follow-up examination.


 > Discussion Top


MALT lymphoma of the tongue is a very rare disease. Head and neck is the second most prevalent site of extranodal lymphoma after the GIT. However, a diffuse large B cell lymphoma is the common histopathology and the Waldeyer's ring, thyroid gland, and salivary glands are its most common sites. [1],[8] MALT lymphoma generally arises in the lymphoid tissue that has been usually acquired as a result of pre-existing chronic inflammatory disorders, such as Helicobacter pylori infection of the stomach. Despite the fact that, MALT is normally present, such as in the tonsil or the Peyer's patches, the incidence of MALT lymphoma is very low. [1],[2],[3]

There are four cases of MALT lymphoma of tongue that have been reported around the world to our knowledge. [4],[5],[6],[7] One case reported of a 43-year-old man with a 1.9 × 1.0 cm tongue tumor. He underwent local resection and he is alive without recurrences at 6 months. [6] Another case is of a 62-year-old woman who was treated with conservative care only. The lymphoma spread to her orbital cavity and bone marrow after 5 years. She was treated with chemotherapy after 7 years of diagnosis and was alive with her disease in good condition 10 years later. [4] The other cases were of a 61 and 80-year-old women, both of whom also underwent only surgical resections and had no recurrences. [5],[7]

Our report concerns the case of a young woman with a past history with peripheral T cell lymphoma in the head and neck region. Although the site of involvement was close and there are some reports of peripheral T cell lymphoma that can mimic the extranodal marginal zone B cell lymphoma, the pathology of the two sites showed definitely different morphologies and immunohistochemical results. [9] MALT lymphomas tend to remain localized for prolonged periods and patients commonly present with a long history. [10] So local therapy alone, either surgery or radiotherapy, may be curative. [11] Deinbeck et al., [12] treated 30 marginal zone lymphoma patients with median 40 Gy of radiation. Ninety percentage of these patients had extranodal presentation, and there was no infield recurrence after median 103 months follow-up.

However, there are also some reports that showed extraintestinal MALT lymphomas had more probability of dissemination and multiorgan involvement. [10] Ueda et al., reported nongastric MALT lymphoma patients had worse progression free survival than gastric MALT lymphoma, even when treated with rituximab-containing chemotherapy. [13]

So whether or not our treatment, radical radiation therapy on the tumor and regional lymphatics, was adequate is controversial. We treated the patient with radiation therapy alone without chemotherapy because she already had a past history of CHOP chemotherapy. We included the entire neck lymphatics because the base of tongue is a site of abundant lymphatics and have a high probability of lymph node metastasis. Since it is not known whether or not our treatment was enough, there needs to be a more long-term follow-up. Nevertheless, we expect that the treatment outcome would be favorable because the previous reports of the MALT lymphoma of tongue treated by surgery alone have shown good long-term results and our patient acquired complete remission early, which has lasted for more than 3 years.

 
 > References Top

1.Zucca E, Conconi A, Pedrinis E, Cortelazzo S, Motta T, Gospodarowicz MK, et al. Nongastric marginal zone B-cell lymphoma of mucosa-associated lymphoid tissue. Blood 2003;101:2489-95.  Back to cited text no. 1
    
2.National Cancer Institute sponsored study of classifications of non-Hodgkin′s lymphomas: Summary and description of a working formulation for clinical usage. The Non-Hodgkin′s Lymphoma Pathologic Classification Project. Cancer 1982;49:2112-35.  Back to cited text no. 2
[PUBMED]    
3.Harris NL, Jaffe ES, Stein H, Banks PM, Chan JK, Cleary ML, et al. A revised European-American classification of lymphoid neoplasms: A proposal from the International Lymphoma Study Group. Blood 1994;84:1361-92.  Back to cited text no. 3
[PUBMED]    
4.Ferry JA, Yang WI, Zukerberg LR, Wotherspoon AC, Arnold A, Harris NL. CD5 extranodal marginal zone B-cell (MALT) lymphoma. A low grade neoplasm with a propensity for bone marrow involvement and relapse. Am J Clin Pathol 1996;105:31-7.  Back to cited text no. 4
    
5.Sakabe H, Bamba M, Nomura K, Kitamura S, Segawa H, Yasui H, et al. MALT lymphoma at the base of the tongue developing without any background of immunodeficiency or autoimmune disease. Leuk Lymphoma 2003;44:875-8.  Back to cited text no. 5
    
6.Kuramoto T, Yao M, Ishikura S, Kano. A case of mucosa-associated lymphoid tissue lymphoma at the tongue. Kokubyo Gakkai Zasshi 1995;44:557.  Back to cited text no. 6
    
7.Goteri G, Ascani G, Filosa A, Rubini C, Olay S, Balercia P. Primary malt lymphoma of the tongue. Med Oral Patol Oral Cir Bucal 2004;9:461-3.  Back to cited text no. 7
    
8.Lim JH, Lim JY, Kim YM, Kim CS, Choi SJ, Yi HG, et al. Primary diffuse large B cell lymphoma of the base of tongue. J Cancer Res Ther 2012;8:135-7.  Back to cited text no. 8
    
9.Uherova P, Ross CW, Finn WG, Singleton TP, Kansal R, Schnitzer B. Peripheral T-cell lymphoma mimicking marginal zone B-cell lymphoma. Mod Pathol 2002;15:420-5.  Back to cited text no. 9
    
10.Thieblemont C, Berger F, Dumontet C, Moullet I, Bouafia F, Felman P, et al. Mucosa-associated lymphoid tissue lymphoma is a disseminated disease in one third of 158 patients analyzed. Blood 2000;95:802-6.  Back to cited text no. 10
    
11.Tsang RW, Gospodarowicz MK, Pintilie M, Wells W, Hodgson DC, Sun A, et al. Localized mucosa-associated lymphoid tissue lymphoma treated with radiation therapy has excellent clinical outcome. J Clin Oncol 2003;21:4157-64.  Back to cited text no. 11
    
12.Deinbeck K, Geinitz H, Haller B, Fakhrian K. Radiotherapy in marginal zone lymphoma. Radiat Oncol 2013;8:2.  Back to cited text no. 12
    
13.Ueda K, Terui Y, Yokoyama M, Sakajiri S, Nishimura N, Tsuyama N, et al. Non-gastric advanced mucosa-associated lymphoid tissue (MALT) lymphoma has worse prognosis than gastric MALT lymphoma even when treated with rituximab-containing chemotherapy. Leuk Lymphoma 2013.  Back to cited text no. 13
    


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



 

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