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An Official Publication of the Indian Association of Oral and Maxillofacial Pathologists


 
  Table of Contents    
CASE REPORT  
Year : 2015  |  Volume : 19  |  Issue : 2  |  Page : 255-259
 

Ill-fitting dentures as primary presentation of mantle cell lymphoma: A case report and literature review of the primary mantle cell lymphomas of the hard palate


1 Department of Oral and Maxillofacial Surgery, Faculty of Dentistry, Eski?ehir Osmangazi University, Eskişehir, Turkey
2 Department of Pathology, Eskişehir Osmangazi University, Eskişehir, Turkey
3 Department of Hematology, Faculty of Medicine, Eskişehir Osmangazi University, Eskişehir, Turkey

Date of Submission13-Oct-2014
Date of Acceptance27-Jul-2015
Date of Web Publication4-Sep-2015

Correspondence Address:
Ömür Dereci
Department of Oral and Maxillofacial Surgery, Faculty of Dentistry, Eskişehir Osmangazi University, Meşelik Campus, 26480 Eskişehir
Turkey
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0973-029X.164545

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   Abstract 

Mantle cell lymphoma (MCL) is a subtype of B-cell non-Hodgkin's lymphoma seen predominantly in males. Common extra-nodal sites of involvement of MCL are Waldeyer's ring, gastrointestinal tract, bone marrow and peripheral blood. The extra-nodal palatal localization of MCL is quite uncommon. MCL is seen in predominantly older patients, therefore undiagnosed MCL patients are likely to have total prosthesis. In this study, a case of MCL, initially presenting as palatal swelling was reported with relevant literature review and the possible role of dental professionals in the diagnosis of this rare entity was discussed.


Keywords: Hard palate, mantle cell lymphoma, non-Hodgkin lymphoma, oral lymphoma


How to cite this article:
Dereci Ö, Ay S, Açıkalın MF, Karagülle M. Ill-fitting dentures as primary presentation of mantle cell lymphoma: A case report and literature review of the primary mantle cell lymphomas of the hard palate. J Oral Maxillofac Pathol 2015;19:255-9

How to cite this URL:
Dereci Ö, Ay S, Açıkalın MF, Karagülle M. Ill-fitting dentures as primary presentation of mantle cell lymphoma: A case report and literature review of the primary mantle cell lymphomas of the hard palate. J Oral Maxillofac Pathol [serial online] 2015 [cited 2019 Oct 23];19:255-9. Available from: http://www.jomfp.in/text.asp?2015/19/2/255/164545



   Introduction Top


Mantle cell lymphoma (MCL) is a B-cell malignant lymphoid tumor which is defined as a separate entity and included in lymphoma classification by World Health Organization (WHO). [1] MCL includes small-medium sized lymphoid cells and accounts for 6-10% of all B-cell lymphomas. [2],[3]

MCL patients are predominantly older males and usually present as stage IV (Ann Arbor) disease. The prognosis of MCL is reported to be poor with a mean survival of 3 years. [1],[4],[5]

In most of the cases the primary presentation site is the lymph nodes. However, extra-nodal sites of involvement such as oral cavity, gastrointestinal tract, Waldeyer's ring, peripheral blood are well documented. [6] Primary hard palate involvement of MCL is reported several times. [7],[8],[9],[10],[11],[12],[13],[14],[15],[16] Palatal MCL is mostly seen in elderly people and may be masked with the presence of prosthesis. In this study, a case of palatal MCL is presented with treatment outcome and a literature analysis was performed to analyze the importance of dental examination in the primary diagnosis of the disease.


   Case report Top


A 71-year-old male patient was referred to Eskişehir Osmangazi University, Faculty of Dentistry, Department of Oral and Maxillofacial Surgery with a complaint of ill-fitting dentures. An intra-oral examination was performed and symmetric mucosal enlargements were observed on hard palate [Figure 1]. The mucosal swellings were asymptomatic. There were no pathologic findings on radiological examination. Patient was uncomfortable about the mobility of his dentures and had tried to fix them by sticking dish rags or other foreign materials into the prosthesis. Medical history was insignificant. Therefore, a provisional diagnosis of reactive mucosal lesion was made and incisional biopsy was performed under local anesthesia.
Figure 1: Ulcerated palatal symmetric mucosal swelling responsible for the mobility of maxillary dentures

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Microscopic examination revealed diffuse abundant small cells with hyperchromatic nuclei in the subepithelial region [Figure 2]. Mucosal ulceration was evident. Mild nuclear enlargement and pleomorphism were observed in small cells [Figure 3]. Immunohistochemistry was performed for differential diagnosis.
Figure 2: Diffuse small cell lymphocytic infiltration is the predominant component on histology. Glandular structures can be seen at the upper-right quadrant (H&E stain, ×100)

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Figure 3: Nuclei of small cells have distinct hyperchromatism. Mild nuclear pleomorphism is also evident (H&E stain, ×400)

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After the immunohistochemistry, tumoral cells showed strong positive staining with CD20 (L26, Ventana, Tucson) [Figure 4], pax-5 (SP34, Ventana, Tucson), CD5 (SP19, Ventana, Tucson), cyclin D1 (SP4-R, Ventana, Tucson) [Figure 5], bcl-2 (124, Ventana, Tucson) and IgM (poliklnal, Ventana, Tucson). Tumoral cells were partially stained with CD43 (L60, Ventana, Tucson). CD21 (SP104, Ventana, Tucson) and CD23 (SP23, Ventana, Tucson) immunostaining showed follicular dendritic cells. CD10 (SP67, Ventana, Tucson) and IgD (policlonal, Ventana, Tucson) were negative. Ki-67 proliferation index was 50%. The case was diagnosed as MCL with these findings. Patient was referred to Hematology Department of Eskişehir Osmangazi University, Faculty of Medicine for further evaluation and treatment.
Figure 4: Tumoral cells stained diffusely positive with CD20 (IHC stain, ×200)

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Figure 5: Tumoral cells showed nuclear positivity with cyclin D1 staining (IHC stain, ×200)

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Ultrasonography of abdomen revealed multiple lymphoadenopathies at mesenteric and para-aortic regions. After informed consent was taken, patient underwent cyclophosphamide, oncovin, prednol (COP) chemotherapy regimen. However, remission was not observed and the patient died after four chemotherapy cycles.


   Materials and methods Top


English dental and medical literature search was conducted using the combination of terms such as hard palate, MCL, lymphoma and extra-nodal lymphoma in PubMed. Cases which were reported under the strict diagnosis of MCL and primarily located on the hard palate were extracted and data regarding treatment, follow-up and demographics was reviewed. Cases with secondary involvement were excluded from the study.


   Results Top


Nine publications with 14 cases defining primary hard palate MCLs published between 1990 and 2014 were identified. The mean age of earlier published cases was 67.1 years. Data regarding demographics and treatment of published cases are shown in [Table 1].
Table 1: Demographic and follow-up data of previously reported cases


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   Discussion Top


MCL is a counterpart of non-Hodgkin lymphoma family and was first included in lymphoma classification in 2001. [1] It originates from peripheral B-cells of mantle zone. [10] MCL is relatively uncommon and occurs in middle aged adults with a male predilection. [1],[2],[10]

Although MCL is mainly located in the lymph nodes, involvement of extra-nodal sites such as Waldeyer's ring, peripheral blood, bone marrow and gastrointestinal tract are also reported. [6]

Definite diagnosis of MCL is achieved with immunohistochemical staining with or without the assistance of molecular techniques. MCL tumoral cells commonly express CD20, CD5, CD43 antigens and BCL2 and cyclin D1 protein. However, they are CD10 and BCL6 negative and show negativity/weak positivity for CD23 antigen expression. [1] Histologic examination reveals abundant monomorphic small-medium sized lymphoid cells with slightly/markedly irregular nuclear contours. [10] The first impression on histologic appearance reminds of small cell lymphoma. Blastoid and pleomorphic subtypes of MCL have been defined by WHO and it is suggested that blastoid or pleomorphic morphology are aggressive variants; similar histology may be seen in some cases at relapse. [18]

Hard palate is an unusual site for primary MCL and this has been reported in the English literature several times. [7],[8],[10],[11],[12],[13],[17] Kyo et al. [7] reported that long term use of maxillary prosthesis might have provoked the lymphoid tissue accumulation and proliferation leading to a mass of MCL in the submucosal region of the hard palate. Guggisberg and Jordan [10] reported that most oral MCLs occur in an elderly male population and have a predilection for the palate. The duration of the prosthesis usage of MCL patients could not be analyzed in the current literature review due to the unavailable data in the case reports. In the current case, patient was using total prosthesis for 10 years and the only complaint of the patient was the mobility of his dentures. The clinical appearance of the tumor suggested that the palatal accumulation of lymphoid mass might have been initiated by the long term use of the total prosthesis.

Other lymphoid tumors may primarily infiltrate hard palate similar to MCL. Milgrom and Yahalom reported nine cases of primary indolent lymphomas of the hard palate including MCL, follicular lymphoma and marginal zone lymphoma. [11] There were two cases of MCL in their series and it is suggested that MCL cases showed more aggressive behavior compared to other lymphomas of the hard palate. [11]

Polychemotherapy is mainly used to treat aggressive non-Hodgkin lymphomas including MCL. (CHOP; cyclophosphamide, doxorubicin, vincristine, prednisolone [19] ) Chang et al. [8] reported a polychemotherapy regimen consisting of Cytoxan, doxorubicin, vincristine and prednisone, in conjunction with rituximab administered every 3 weeks. Different chemotherapeutics achieve approximately high treatment response rate of 70%. [20],[21],[22] Meusers et al. [23] reported that the CHOP regimen did not show superiority to COP regimen in the treatment of MCL. Although CHOP-like antracycline containing chemotherapeutic combinations do not have distinct advantage on survival, they are currently chosen for standard therapuetic approach. [3] Radiation therapy is still a therapeutic option in low-stage disease (Ann-Arbor stage 1,2) In advance stage disease, the combination of radiotherapy and chemotherapy can be used, however, the efficacy of this combination have not been proven yet.


   Conclusion Top


Primary MCL of the hard palate is a rare entity which is mostly seen in elder people. In this study, a case of palatal primary MCL which presents with ill-fitting total prosthesis is reported with clinical and histopathological features. Extensive literature review revealed 14 previously reported cases. Dental total prosthesis may mask the existing lesion on the hard palate, therefore, dental practitioner should be alert at the time of the clinical examination of patients with total or partial prosthesis.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
   References Top

1.
Harris NL, editor. WHO Classification of Tumours of Haematopoietic and Lymphoid Tissues. Lyon: IARC Press: 2001.  Back to cited text no. 1
    
2.
Zhou Y, Wang H, Fang W, Romaguer JE, Zhang Y, Delasalle KB, et al. Incidence trends of mantle cell lymphoma in the United States between 1992 and 2004. Cancer 2008;113:791-8.  Back to cited text no. 2
    
3.
Lenz G, Dreyling M, Hiddemann W. Mantle cell lymphoma: Established therapeutic options and future directions. Ann Hematol 2004;83:71-7.  Back to cited text no. 3
    
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Hoster E, Dreyling M, Klapper W, Gisselbrecht C, van Hoof A, Kluin-Nelemans HC, et al. A new prognostic index (MIPI) for patients with advanced-stage mantle cell lymphoma. Blood 2008;111:558-65.  Back to cited text no. 4
    
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Tiemann M, Schrader C, Klapper W, Dreyling MH, Campo E, Norton A, et al. Histopathology, cell proliferation indices and clinical outcome in 304 patients with mantle cell lymphoma (MCL): A clinicopathological study from the European MCL Network. Br J Haematol 2005;131:29-38.  Back to cited text no. 5
    
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Swerdlow SH, Campo E, Seto M, Müller-Hermelink HK. Mantle cell lymphoma. In: Swerdlow SH, Campo E, Harris NL, Jaffe ES, Pileri SA, Stein H, et al., editors. WHO Classification of Tumours of Haematopoietic and Lymphoid Tissues. Lyon: IARC; 2008. p. 229-32.  Back to cited text no. 6
    
7.
Kyo C, Kawaoka Y, Kinoshita K, Ohno H. Mantle cell lymphoma presenting with a tumor of the hard palate. Intern Med 2010;49:1663-6.  Back to cited text no. 7
    
8.
Chang CC, Rowe JJ, Hawkins P, Sadeghi EM. Mantle cell lymphoma of the hard palate: A case report and review of the differential diagnosis based on the histomorphology and immunophenotyping pattern. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2003;96:316-20.  Back to cited text no. 8
    
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Triantafillidou K, Dimitrakopoulos J, Iordanidis F, Gkagkalis A. Extranodal non-hodgkin lymphomas of the oral cavity and maxillofacial region: A clinical study of 58 cases and review of the literature. J Oral Maxillofac Surg 2012;70:2776-85.  Back to cited text no. 9
    
10.
Guggisberg K, Jordan RC. Mantle cell lymphoma of the oral cavity: Case series and comprehensive review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2010;109:98-104.  Back to cited text no. 10
    
11.
Milgrom SA, Yahalom J. Indolent non-Hodgkin lymphoma primarily involving the hard palate: Outcome following radiotherapy. Leuk Lymphoma 2013;54:1208-11.  Back to cited text no. 11
    
12.
Scheller K, Becker S, Scheller C. Symmetric palatal swelling as the first clinical manifestation of a mantle cell non-Hodgkin's lymphoma: A case report and review of literature. J Oral Maxillofac Pathol 2011;15:311-5.  Back to cited text no. 12
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13.
Aguilera NS, Bijwaard KE, Duncan B, Krafft AE, Chu WS, Abbondanzo SL, et al. Differential expression of cyclin D1 in mantle cell lymphoma and other non-Hodgkin's lymphomas. Am J Pathol 1998;153:1969-76.  Back to cited text no. 13
    
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Tan KB, Tan LH, Soo R, Putti TC, Chong SM. Involvement of the appendix and palate by pleomorphic variant mantle cell lymphoma. Leuk Lymphoma 2006;47:1704-7.  Back to cited text no. 14
[PUBMED]    
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Fitzpatrick SG, Al-Quran SZ, Fetner AE, Kuklan RM, Cohen DM, Bhattacharyya I. Mantle cell lymphoma of the hard palate: A series of 4 cases. Oral Surg 2012;5:76-83.  Back to cited text no. 15
    
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Costa FW, Chaves FN, Pereira KM, Alves AP, Sousa FB, Bêco MP, et al. Initial presentation of mantle cell lymphoma in the hard palate. Oral Surg Oral Med Oral Pathol Oral Radiol 2014;117:e66.  Back to cited text no. 16
    
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Kolokotronis A, Konstantinou N, Christakis I, Papadimitriou P, Matiakis A, Zaraboukas T, et al. Localized B-cell non-Hodgkin's lymphoma of oral cavity and maxillofacial region: A clinical study. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2005;99:303-10.  Back to cited text no. 17
    
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Banks PM, Chan J, Cleary ML, Delsol G, De Wolf-Peeters C, Gatter K, et al. Mantle cell lymphoma. A proposal for unification of morphologic, immunologic, and molecular data. Am J Surg Pathol 1992;16:637-40.  Back to cited text no. 18
    
19.
van der Waal RI, Huijgens PC, van der Valk P, van der Waal I. Characteristics of 40 primary extranodal non-Hodgkin lymphomas of the oral cavity in perspective of the new WHO classification and the International Prognostic Index. Int J Oral Maxillofac Surg 2005;34:391-5.  Back to cited text no. 19
    
20.
Meusers P, Hense J, Brittinger G. Mantle cell lymphoma: Diagnostic criteria, clinical aspects and therapeutic problems. Leukemia 1997;11 Suppl 2:S60-4.  Back to cited text no. 20
    
21.
Vandenberghe E, De Wolf-Peeters C, Vaughan Hudson G, Vaughan Hudson B, Pittaluga S, Anderson L, et al. The clinical outcome of 65 cases of mantle cell lymphoma initially treated with non-intensive therapy by the British National Lymphoma Investigation Group. Br J Haematol 1997;99:842-7.  Back to cited text no. 21
    
22.
Zucca E, Roggero E, Pinotti G, Pedrinis E, Cappella C, Venco A, et al. Patterns of survival in mantle cell lymphoma. Ann Oncol 1995;6:257-62.  Back to cited text no. 22
    
23.
Meusers P, Engelhard M, Bartels H, Binder T, Fülle HH, Görg K, et al. Multicentre randomized therapeutic trial for advanced centrocytic lymphoma: Anthracycline does not improve the prognosis. Hematol Oncol 1989;7:365-80.  Back to cited text no. 23
    


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