Mantle cell lymphoma of the oral cavity: case series and comprehensive review of the literature

doi:10.1016/j.tripleo.2009.08.010

Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology, and Endodontology

Volume 109, Issue 1, January 2010, Pages 98-104

https://doi.org/10.1016/j.tripleo.2009.08.010 Get rights and content

Objective

Mantle cell lymphoma (MCL) is a rare B-cell neoplasm that has only recently been defined as a distinct entity. Because of its rarity and histologic similarities to other small cell lymphomas, the microscopic diagnosis of MCL may be challenging. This is particularly true within the oral cavity, where other lymphomas are more frequent. To date, few cases of MCL presenting within the oral cavity have been reported.

Study design

We present 2 new cases of MCL within the oral cavity and systematically review 7 other cases of MCL reported in the English-language literature. Historical cases were reviewed, and available data regarding morphology, special stains, demographics, clinical presentation, radiographic findings, management, and outcome were extracted. Data from our present series were then compared with the earlier published literature.

Results

To the best of our knowledge, this is the largest reviewed series of MCL within the oral cavity, totaling 9 cases. The features of our cases, including histology, clinical presentation, and outcome, are consistent with the 7 earlier reported cases. The majority of oral MCLs occur in an older male population, and a high proportion occur on the palate.

Conclusion

We conclude that MCL of the oral cavity is an uncommon diagnosis. Most oral MCLs occur in an elderly male population and have a possible predilection for the palate. The microscopic diagnosis can be challenging, given its similar appearance to other small cell lymphomas, requiring a comprehensive immunohistochemical panel for the accurate diagnosis. Like MCL occurring in other sites in the body, the prognosis and outcome of oral MCL appears to be poor.

Section snippets

Case series

The databases of the University of California San Francisco Oral Pathology Biopsy Service and Department of Pathology were systematically searched for all MCLs occurring within the oral cavity for the period 2001-2007. All slides and case reports were retrieved and reviewed. For cases reported before 2001, when the term mantle cell lymphoma was not commonly used, the databases were searched for all lymphomas occurring in the oral cavity and then the original sections and case reports were

Results

We identified 2 oral cavity MCL cases from within our files (Table II) and an additional 7 published cases. All cases diagnosed as small cell lymphomas of the oral cavity before 2001 were reviewed (n = 70). Of these 70 cases, 12 were subsequently stained with CCND1. All of these failed to demonstrate CCND1 staining. The clinical findings of all 9 patients are summarized in Table II. The mean patient age of our 2 patients at presentation was 77 years, similar to the overall mean age of 71 years

Discussion

Although as a group, NHL is the second most common malignancy presenting within the head and neck region, involvement of the head and neck by MCL is unusual, and the primary presentation of MCL within the oral cavity is rare.3, 4, 6, 7, 8, 9, 10, 11, 12, 13, 14, 15, 16 To our knowledge, this is the largest reviewed series of MCL arising within the oral cavity that has been reported in the English-language literature. The pathologic and demographic findings of our 2 patients are generally

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Extranodal manifestation of mantle cell lymphoma in the submandibular duct: A case report and review of literature
2023, Otolaryngology Case Reports
Citation Excerpt :
Despite its indolent course, the prognosis in patients with MCL is poor. In a series of 9 cases of oral cavity MCL by Guggisberg and Jordan [10], mean survival was only 3 years, though prognosis correlated directly with stage at diagnosis, age, and present comorbidities. Thus, regardless of clinical acuity, timely diagnosis and referral to the appropriate specialists is the cornerstone of treatment.
The vast majority of oral cavity malignancies are primary squamous cell carcinomas. Far less common are lymphomas, sarcomas, and secondary cancers. We present an interesting case of a patient with relapsed lymphoma that presented as a submandibular duct-occupying, elongated mass on the floor of mouth, similar in presentation to a massive sialolith. Although metastasis to the oral cavity is rare, consideration of this possibility is necessary in the evaluation of a patient with prior malignancy. The diligent clinician should be aware that these oral metastases can resemble common and innocuous lesions, which may delay accurate diagnosis. Timely biopsy and proper utilization of specialized testing such as IHC and flow cytometry are crucial to early initiation of therapy and optimization of care.
Mantle cell lymphoma involving the oral and maxillofacial region: a study of 20 cases
2023, Oral Surgery, Oral Medicine, Oral Pathology and Oral Radiology
Citation Excerpt :
Males predominate in most of the reported series,1,15 a feature also documented in the current sample. Cases involving the oral cavity usually present as asymptomatic nonulcerated swellings, with the palate being the most affected subsite.10,16,17 Two of our cases presented as diffuse bilateral swellings, 1 of which also showed simultaneous involvement of the upper lip.
To investigate the clinicopathologic features of mantle cell lymphoma (MCL) involving the oral and maxillofacial region.
The MCL cases were retrieved from the pathosis database of 6 pathology laboratories. Original hematoxylin and eosin slides and immunohistochemical reactions were reviewed for confirmation of the initial diagnosis. Clinical data of the cases were obtained from the patients’ pathosis and/or medical charts.
Twenty cases were included in the study, showing a male predominance and a mean age of 66 years. The oral cavity (12 cases) and the oropharynx (5 cases) were the most commonly involved subsites. Most cases presented as asymptomatic swellings, with 2 cases showing bilateral involvement of the palate. The classic histologic variant predominated (12/20 cases). All cases expressed CD20 with nuclear cyclin D1 positivity. SOX11 was seen in 9/13 cases, CD5 in 6/16 cases, Bcl2 in 16/19 cases, CD10 in 2/20 cases, and Bcl6 in 4/16 cases. Ki67 showed a mean proliferation index of 40.6%. The Epstein-Barr virus (EBV) was negative in all cases investigated. Follow-up data was available for 7 patients, with 5 currently alive and 2 deceased.
Mantle cell lymphoma, albeit rare, may manifest in the oral and maxillofacial region. Its histologic heterogeneity demands a high degree of diagnostic skill from pathologists.
Mantle cell lymphoma involving tonsils: a clinicopathologic study of 83 cases
2021, Human Pathology
Citation Excerpt :
Involvement of Waldeyer's ring is known to be seen in about 5–10% of patients with MCL. However, the available literature is mostly case reports and small case series and there are very few studies focused on MCL involving the tonsil [11–18]. In this study, we describe the clinicopathologic features and prognosis of patients with MCL with involvement of the tonsils.
We report 83 cases of mantle cell lymphoma (MCL) involving the tonsil as initial manifestation (IM). The median age at the time of tonsillar involvement was 58 years (range, 35–79 years). Most (85%) patients presented similar to acute tonsillitis. Lymphadenopathy (84%) and advanced stage of disease (81%) were frequent. With a median follow-up of 6.1 years (range, 0.5–18.4 years), the median overall survival (OS) was 11.3 years for all patients. Cases with classic MCL morphology demonstrated a superior OS (median OS: 11.7 years versus 7.8 years for aggressive morphology, P = 0.0361). Approximately 20% of patients had limited stage of disease, and they had excellent outcomes (median OS: not reached versus 11.3 years for advanced-stage MCL, P = 0.0479). All the patients were alive after a median follow-up of 6.6 years (range, 1–16.2 years). There were no differences in relapse-free survival in morphology and stage (P > 0.05). When tonsils were involved by relapsed MCL, patients less commonly had acute tonsillitis–like symptoms, lymphadenopathy, and advanced stage of disease compared to MCL as IM. Patients in the relapse group had poorer OS than patients in the IM group from the time of tonsillar involvement by MCL to the date of death or last follow-up (7.8 versus 11.7 years, P = 0.003). Compared with a group of 93 patients whose initial biopsy specimen was a lymph node, patients whose initial biopsy specimen was tonsil had similar OS (11.7 versus 8.8 years, P = 0.1764). However, patients with tonsillar MCL more commonly had limited stage disease (19% versus 8%, P = 0.0385) and a low-risk Mantle Cell Lymphoma International Prognostic Index score (71% versus 47%, P = 0.0025).
Mantle cell lymphoma of the oral cavity: An uncommon site for an uncommon lesion, two new cases and literature review
2020, Oral Oncology
Mantle cell lymphoma of palatine tonsil: A case report
2020, Respiratory Medicine Case Reports
Citation Excerpt :
Primary MCL of palatine to nsil is rare. Extensive review of English literature review revealed 17 previously reported cases [3–6]. We report a rare case of primary MCL of the palatine tonsil.
Mantle cell lymphoma presenting as a symmetrically hard palatal swelling: A case report and review of the literature
2015, Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology
Mantle cell lymphoma (MCL) is a rare B-cell neoplasm accounting for approximately 3–10% of all non-Hodgkin lymphomas (NHLs). Initial clinical manifestation in the oral cavity is extremely rare. We report a case of MCL involving the hard palate. The patient, a 92-year-old man, presented at the department of oral surgery of our hospital because of swelling of the palate, causing instability of his maxillary denture. On examination, the hard palate was symmetrically swollen, and computed tomography revealed an extensive soft tissue mass with osteolysis. Biopsy of the mass disclosed monotonous diffuse infiltrates of small to intermediate-sized lymphoid cells underneath the squamous epithelium. Immunohistochemistry revealed that the cells were positive for CD5, CD20, CD79a, bcl-2, cyclin D1, ki-67 (MIB-1 index, 68.5%) and (weakly or strongly) CD43, and negative for CD3, CD10, CD21, CD23, bcl-6, TdT and EBV-latent infection membrane protein-1. In situ hybridization also showed negativity for EBV-encoded small RNA. These findings were histologically compatible with MCL. To our knowledge, this is the first reported case of MCL initially manifesting as a symmetrical mass on the hard palate with osteolysis in such an elderly patient.

View all citing articles on Scopus

: Supported by the National Institutes of Health (CA095231, T32DE017249, and T32DE019096).

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Oral and maxillofacial pathologyMantle cell lymphoma of the oral cavity: case series and comprehensive review of the literature

Objective

Study design

Results

Conclusion

Section snippets

Case series

Results

Discussion

Ann Oncol

Oral Surg Oral Med Oral Pathol Oral Radiol Endod

Clin Oncol (R Coll Radiol)

Int J Oral Maxillofac Surg

Hum Pathol

Ann Oncol

Blood

Blood

Oral Surg Oral Med Oral Pathol Oral Radiol Endod

Oral Surg Oral Med Oral Pathol Oral Radiol Endod

Blood

Mantle cell lymphomaA proposal for unification of morphologic, immunologic, and molecular data

Am J Surg Pathol

Incidence trends of mantle cell lymphoma in the United States between 1992 and 2004

Cancer

Oral and maxillofacial pathology
Mantle cell lymphoma of the oral cavity: case series and comprehensive review of the literature