Solitary plasmacytoma of maxilla: Rare case report

Sandhya Sundar; Pratibha Ramani; Gheena Ranjith; Abilasha Ramasubramanian

doi:10.4103/ijhi.IJHI_4_22

CASE REPORT

Year : 2019 | Volume : 8 | Issue : 1 | Page : 16-18

Solitary plasmacytoma of maxilla: Rare case report

Sandhya Sundar, Pratibha Ramani, Gheena Ranjith, Abilasha Ramasubramanian
Department of Oral Pathology, Saveetha Dental College & Hospitals, Saveetha Institute of Medical and Technical Sciences, Chennai, Tamil Nadu, India

Date of Submission	07-Feb-2022
Date of Decision	08-Feb-2022
Date of Acceptance	08-Feb-2022
Date of Web Publication	22-Apr-2022

Correspondence Address:
Sandhya Sundar
Department of Oral Pathology, Saveetha Dental College & Hospitals, Saveetha Institute of Medical and Technical Sciences, 9 Cholapuram Street, Thiruvanmiyur, Chennai 600041, Tamil Nadu
India

Source of Support: None, Conflict of Interest: None

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DOI: 10.4103/ijhi.IJHI_4_22

Abstract

Solitary plasmacytomas are rare group of neoplasms accounting for less than 5% of plasma cell dyscrasia. Here in, we report an unusual case of solitary plasma cell neoplasm in the maxilla in a 43-year-old female patient. Careful clinical and pathological evaluation revealed the isolated tumor of the plasma cells with absence of multiple bones and other extra-oral tissue involvement. As this isolated lesion has increased propensities to transform to its systemic counterpart, malignant melanoma—a constant, periodic follow-up is necessitated.

Keywords: Cart wheel pathology, multiple myeloma, plasmacytoma, plasma cell dyscrasia, plasma cell tumor, serum M component

How to cite this article:
Sundar S, Ramani P, Ranjith G, Ramasubramanian A. Solitary plasmacytoma of maxilla: Rare case report. Int J Histopathol Interpret 2019;8:16-8

How to cite this URL:
Sundar S, Ramani P, Ranjith G, Ramasubramanian A. Solitary plasmacytoma of maxilla: Rare case report. Int J Histopathol Interpret [serial online] 2019 [cited 2023 Dec 7];8:16-8. Available from: https://www.ijhi.org/text.asp?2019/8/1/16/343727

Introduction

Plasma cell neoplasms form a spectrum of rare disorders. They include monoclonal gammopathy of undetermined significance, isolated plasmacytoma of the bone, extra medullary plasmacytoma, and multiple myeloma.^[1] Proliferation of single B lymphocyte clone with secretion of single homogeneous immunoglobulin or its fragments (serum M component) is characteristically associated in these lesions.

Case Report

A 43-year-old woman came to the college with a chief complaint of swelling in the left posterior part of the maxillary region. The duration was 1 month. History revealed that she had undergone restoration in the left upper back tooth region of the jaw a year back and extraction of a tooth in the same region. There was no relevant medical, surgical, or personal history.

General examination did not reveal any abnormality or deformity. The patient had presented with moderately nourished and built stature. There was no evidence of systemic derangement. Extraorally, there was presence of facial asymmetry in the left side with normal mouth opening and competent lips. No palpable lymph nodes were identified. Intraorally, there was a single swelling measuring 4 × 4 cm in the left maxillary alveolus extending from distal of 25 to the maxillary tuberosity. On palpation, it was bony hard swelling with surface indentations of opposing teeth and induration.

Orthopantomogram analysis evinced the presence of multilocular radiolucency intervened by the bony trabeculae. There were irregular ragged borders simulating “moth-eaten” appearance. The lesion was extending into the maxillary sinus space. The computed tomography scan of the area showed an erosive lesion succumbing the whole of the left maxillary area almost reaching to the base of the skull.

Based on the grounds of clinical and pathological consensus, a provisional diagnosis of “neoplastic swelling of maxilla” was given with differentials of ameloblastoma, fibro-osseous lesion, or myxoma. The aspiration attempted was negative ruling out the possibility of any cystic lesion. Incisional biopsy procedure was done with gross features of two soft tissue bits approximately measuring 1 × 0.5 cm.

On histopathological examination of the specimen [Figure 1], there revealed a highly cellular area deep inside the connective tissue stroma. The superficial epithelium was apparently normal. The cells in the dense area were isomorphic and round to oval shaped with eosinophilic cytoplasm and eccentrically placed nucleus resembling plasma cells. Intracellular areas of perinuclear clearing and areas of extracellular eosinophilic materials were evidenced. The provisional diagnosis of plasmacytoma with differential of multiple myeloma, lymphoplasmacytic lymphoma, or plasmablastic lymphoma was contemplated.

Figure 1: Histopathological picture of the lesion: (a) At 4×, showing the lesion in the deep connective tissue, (b) at 10×, showing the accumulation of the single cell population composed of ovoid cells with eccentric nucleus plasma cells, and (c and d) at 40×, showing isomorphic, round to oval shaped cells with eosinophilic cytoplasm and eccentrically placed nucleus resembling plasma cells

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The immunohistochemical analysis revealed 70% positivity of the tumor cells for the kappa light chain. The serum analysis had shown blood cells counts in normal limits with normal kidney function. Urine analysis did not infer any abnormality—there was absence of urine Bence Jones protein.

Discussion

Plasma cell neoplasms form a spectrum of rare disorders. The aberrant activation and proliferation of a single B lymphocyte clone synthesize and secrete single homogeneous immunoglobulin or its fragments. The neoplasms falling into the group have the presence of M component (monoclonal immunoglobulin) in the serum and free light chains in the urine analysis (Bence Jones protein).^[2],[3],[4]

The isolated plasma cell masses form the “solitary plasma cell tumor.” It accounts for 3–5% plasma cell neoplasms. In many times, it is considered as the forerunner of disseminated disease. Malignant transformation of a single plasma cell underlies the lesion. Deletion 13q, 1p, and 14q and translocation 14q32 and 4p16 have been implicated in the oncotransformation process. Interleukin 6 and other osteo-destructive cytokines are the principal growth factors associated with the lesion. Combined radiotherapy and chemotherapy gives excellent prognosis.^[5]

Solitary plasmacytoma in oral cavity characteristically affects individuals of 40–60 years (mean age 55 years).Males are predominantly involved (in ratio of 2:1) with the predilection for site of posterior mandible.^[6] Very few reports have evidenced the occurrence of lesions in maxilla.^[7] It presents as painless swelling with lytic lesions. Serum M component is detected in 25% of the cases with the absence of systemic involvement.^[6] In the present case, the patient was a female of 43 years who gave a history of pain and swelling in the posterior maxilla. Expansile lytic lesions were present with positive serum M component. There was no evidence of end organ damage.

Multiple myeloma, systemic counterpart of solitary plasma cell tumor, presents as multifocal, punched-out lytic lesions. It is the most common malignant plasma tumor. It presents with pain and systemic involvement. Serum M component is present in all (100%) of the cases. Thus, it is important to differentiate isolated plasma cell tumors from this lesion, which is elicited by serum, urine analysis (presence of Bence Jones protein), and skeletal survey.^[8],[9]

In all, 65–84% of these solitary lesions progress to multiple myeloma in 10 years time. Kyleet al. have recorded three patterns of treatment failure of solitary plasmacytoma—54% resulting in multiple myeloma, 11% resulting in recurrence, and 2% in new bone lesions.^[10] The determining prognostic features would be age, lesion size, M protein levels, and presence of spine lesions.

Conclusion

The report presents a rare case, which caused the painless volume increase in maxilla. Although commonly presented as multilocular radiolucency imitating other common lesions, such rare possibilities should also be carefully considered. Early diagnosis of such life-threatening lesions is important. If the systemic involvement is ruled out, the treatment of choice is the radiotherapy with the follow-up of at least 3 years.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

References

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