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CASE REPORT
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Solitary extramedullary plasmacytoma: A rare gingival lesion


1 Assistant Professor, Department of Dentistry (Periodontics), Maharaja Suhel Dev Autonomous State Medical College and Mahrishi Balark Hospital, Bahraich, Uttar Pradesh, India
2 Assistant Professor, Department of Pathology, Maharaja Suhel Dev Autonomous State Medical College and Mahrishi Balark Hospital, Bahraich, Uttar Pradesh, India

Date of Submission17-Nov-2017
Date of Decision05-Aug-2020
Date of Acceptance07-Aug-2020

Correspondence Address:
Anshul Sawhney,
Department of Dentistry (Periodontics), Maharaja Suhel Dev Autonomous State Medical College and Mahrishi Balark Hospital, Bahraich - 271 801 Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/mjdrdypu.mjdrdypu_215_17

  Abstract 


Plasma cell tumors consist of extramedullary plasmacytoma (EMP), multiple myeloma (MM), and solitary bone plasmacytoma (SBP). EMP is defined as neoplastic proliferation of plasma cells in soft tissues. It presents as a solitary discrete mass either in soft tissue as EMP or in bone as SBP or as disseminated MM. It is a very rare manifestation. It accounts for <1% of all head-and-neck malignancies with oral cavity being the rarest.

Keywords: Gingiva, neoplastic, plasmacytoma



How to cite this URL:
Sawhney A, Sawhney MR. Solitary extramedullary plasmacytoma: A rare gingival lesion. Med J DY Patil Vidyapeeth [Epub ahead of print] [cited 2021 Feb 26]. Available from: https://www.mjdrdypv.org/preprintarticle.asp?id=308714




  Introduction Top


Extramedullary plasmacytoma (EMP) is defined as neoplastic proliferation in soft tissues. It accounts for up to 3% of all plasma cell tumors. These tumors are four times more common in men, in their sixth to eighth decades of life.[1] Approximately, 90% of EMPs are found in the head-and-neck (H and N) region commonly affecting the nasal cavity, paranasal sinuses, tonsillar fossa, and oral cavity.[1],[2] It accounts for approximately 1%–2% of human malignancies and occurs at a rate of about 3.5/100,000 per year.[3],[4] Etiology of this disease is unknown but chronic stimulation, overdose irradiation, viruses, and gene interaction in the reticuloendothelial system.[5]

Plasmacytomas can be graded as lesions of minimal to severe dysplasia.[6] In this article, we reported an EMP in an otherwise healthy young female.


  Case Report Top


A 25-year-old female patient reported to the Outpatient Department of Dentistry (Periodontology), Maharaja Suhel Dev Autonomous State Medical College and Mahrishi Balark Hospital, Bahraich, with a chief complaint of mass in her upper anterior region for the last 6 months [Figure 1]. The growth was asymptomatic and her only concern was cosmetic. Patient also revealed that the growth was increasing from last 1 month. Size of the gingival growth was 2 cm × 2.5 cm × 3 cm. It was soft, friable, and pedunculated. The growth was extending interdentally between 11 and 21. The lesion was hemorrhagic, and spontaneous bleeding on exploration of lesion was there. There was Grade I and II mobility with respect to 11 and 21, respectively. Midline diastema was evident. On pulp vitality testing, using pulp Tester both 11 and 21 were found to be vital. Medical history was not significant. Intraoral periapical radiograph was taken revealing Grade I bone loss with respect to 11 and 21. Complete blood count, liver function test and kidney function test were done and were normal with no evidence of anemia and hypercalcemia. The surgical procedure was done under local anesthetic after explaining and taking written consent from the patient and her family members.
Figure 1: Patient showing mass in her upper anterior region

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Use of pocket marker was done to mark the course of the incision. Excisional biopsy was done using 11 and 15 number Bard-Parker Stainless Steel Blades, followed by localized gingivectomy using gingivectomy knives. The incision was beveled at approximately 45° and should create normal festooned pattern of gingiva. Incision was started apical to the bleeding points and is directed coronally to the points as close as possible to the bone. Granulation tissue was carefully curetted. Use of electrocautery was also done to control bleeding. The sectioned growth was sent to department of pathology for histopathological examination in 10% phosphate-buffered formalin vial. Patient was given postoperative instructions, prescribed tablet amoxiclav 625 mg Tds, tablet Ketorol DT, and tablet Pantop 40 mg for 5 days. Patient was recalled after 2 weeks for follow-up.

On microscopy, section revealed unremarkable stratified squamous epithelium overlying mass composed of sheets of plasma cells having round, hyperchromatic, eccentrically placed nucleus with moderate amount of eosinophilic cytoplasm. Majority of these cells were mature plasma cells [Figure 2]. On immunohistochemistry (IHC), these cells were negative for kappa and positive for CD138 and lambda protein [Figure 3] and [Figure 4]. Bone marrow biopsy failed to show evidence of myeloma. The skeletal survey was also unremarkable. Serum proteins and immunoglobulin levels were within normal limits. Urine examination showed the absence of Bence–Jones protein. Based on histology and IHC, diagnosis of solitary plasmacytoma was made. Tumor cells were negative.
Figure 2: Section shows sheets of plasma cells with eccentric nuclei and moderate amount of eosinophilic cytoplasm (H and E, ×400)

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Figure 3: Section showing negative kappa staining (IHC, ×400)

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Figure 4: Section showing positive brown staining with lambda (IHC, ×400)

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  Discussion Top


Plasma cells are medium sized round-to-oval cells with eccentrically placed nuclei. The cells range in size from 10 to 20 μm, and the nuclear cytoplasmic ratio is 1:2. The nuclear chromatin is arranged in a cart-wheel or clock-face pattern. Main function of plasma cells is to produce immunoglobulins or antibodies. Plasma cell neoplasm can be classified into the following types: multiple myeloma (MM) (bone marrow and other systemic involvements), solitary myeloma (bone plasmacytoma), extramedullary (soft tissue) plasmacytoma, and plasmablastic sarcoma. Primary plasmacytoma, whether osseous or nonosseous, is distinguished from MM by the absence of hypercalcemia, renal insufficiency and anemia, normal skeletal survey, absence of bone marrow plasmacytosis, and serum or urinary paraprotein <2 g/dl.[5] Primary EMP can be solitary or multiple.[7] EMP comprises <1% of all H and N tumors, in which oral cavity is the least commonly involved site. The differential diagnosis of EMP is plasma cell granuloma, pseudolymphoma, and reactive plasmacytic hyperplasia. EMP affects males three to four times more often than females, with an average age of 55. It occurs mainly in the fifth and sixth decades of life. However, one-third of patients with EMP are under 50 years old. EMP affecting the gingiva as a soft and gelatinous growth in the left upper canine is a very rare. It was first described by Martinelli and Rulli in 1968 as a sessile neoplasm from left-to-right canine, easily to be confused with chronic gingivitis.[8] Moshref et al. reported a polypoidal growth in left upper canine clinically similar to peripheral giant cell granuloma.[9]

Microscopically, EMP should be differentiated from other types of plasma cell tumors. The presence of Dutcher bodies and monoclonal expression of plasma cells on histopathology are helpful in distinguishing plasmacytoma from plasma cell granuloma and reactive plasmacytosis.[10] The cells in plasmablastic lymphoma are more single cell type without maturation and not associated with monoclonal gammopathy as in plasmacytoma.[11]

Histopathologic features of EMP show a connective tissue greatly infiltrated by plasma cells which are such as focal sheets, small islands, or plasmacytoid nodules.[12] The treatment of choice for EMP is radiotherapy because the disease is highly radiosensitive.[13] Mendenhall et al.[3] have reported 94% local control rate in solitary plasmacytoma with doses exceeding 40 GY in 4 weeks, while some other researchers have shown less favorable results.[14],[15] Since these granulomas associated with various dental infections show an increase of plasma cells, extra precaution is required for diagnosing solitary lesions.[16] Management involves local eradication of the lesion.[17] If the surgical margins are involved, the patient should receive adjuvant chemotherapy.[18] In various studies, it has been shown previously that adjuvant chemotherapy is sometimes indicated in an attempt to delay the conversion of the disease to myeloma.[3],[17]

Local recurrence has been reported to be up to 10%.[1] Dissemination of the tumor takes place in 35%–50% of EMPs.[19] The patient was recalled after 2 weeks after surgical excision. She had no complaint and was satisfied with the procedure. Healing was uneventful. Surface epithelialization was almost complete after which he did not turn for follow-up.

Martinelli and Rulli[8] in 1968 reported a case of EMP of the gingiva. Their patient had pain and gingivitis for about 1 year. However, in our case, patient came with complaint of growth from the past 6 months which was asymptomatic and was increasing in size.


  Conclusion Top


We present a rare case of primary solitary EMP involving gingiva. Surgical removal of tumor can be performed as a sole treatment of small masses, even in cases where local irradiation has not been successful in eliminating the mass, as in the case of local deposit of amyloid substance or residual disease.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Majumdar S, Raghavan U, Jones NS. Solitary plasmacytoma and extramedullary plasmacytoma of the paranasal sinuses and soft palate. J Laryngol Otol 2002;116:962-5.  Back to cited text no. 1
    
2.
Neville BW, Damm DD, Allen CM. Oral and Maxillofacial Pathology. Philadelphia: Saunders; 2002. p. 527-8.  Back to cited text no. 2
    
3.
Mendenhall WM, Mendenhall CM, Mendenhall NP. Solitary plasmacytoma of bone and soft tissues. Am J Otolaryngol 2003;24:395-9.  Back to cited text no. 3
    
4.
Nofsinger YC, Mirza N, Rowan PT, Lanza D, Weinstein G. Head and neck manifestations of plasma cell neoplasms. Laryngoscope 1997;107:741-6.  Back to cited text no. 4
    
5.
Pisano JJ, Coupland R, Chen SY, Miller AS. Plasmacytoma of the oral cavity and jaws: A clinicopathologic study of 13 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1997;83:265-71.  Back to cited text no. 5
    
6.
Diebold J. World Health Organization classification of malignant lymphomas. Exp Oncol 2001;23:101-3.  Back to cited text no. 6
    
7.
Galieni P, Cavo M, Pulsoni A, Avisati G, Bigazzi C, Neri S, et al. Clinical outcome of extramedullary plasmacytoma. Hematologica 2000;85:47-51.  Back to cited text no. 7
    
8.
Martinelli C, Rulli MA. Primary plasmacytoma of soft tissue (gingiva). Report of a case. Oral Surg Oral Med Oral Pathol 1968;25:607-9.  Back to cited text no. 8
    
9.
Moshref M, Mashhadi-Abbass F, Sargolzaei S and Nafarzadeh S. Extramedullary plasmacytoma of the gingiva. Arch Iran Med 2007;10:91-3.  Back to cited text no. 9
    
10.
Anila Namboodiripad PC, Jaganath M, Sunitha B, Sumathi A. Plasma cell granuloma in the oral cavity. Oral Surg 2009;1:206-12.  Back to cited text no. 10
    
11.
Vega F, Chang CC, Medeiros LJ, Udden MM, Cho-Vega JH, Lau CC, et al. Plasmablastic lymphomas and plasmablastic plasma cell myelomas have nearly identical immunophenotypic profiles. Mod Pathol 2005;18:806-15.  Back to cited text no. 11
    
12.
Liebross RH, Ha CS, Cox JD, Weber D, Delasalle K, Alexanian R. Clinical course of solitary extramedullary plasmacytoma. Radiother Oncol 1999;52:245-9.  Back to cited text no. 12
    
13.
Rawat DS, Grover M, Verma PC. Extramedullary plasmacytoma of nasal cavity: A rare entity. Clin Rhinol Int J 2010;3:39-41.  Back to cited text no. 13
    
14.
Kapadia SB, Desai U, Cheng VS. Extramedullar y plasmacytoma of the head and neck. A clinicopathologic study of 20 cases. Medicine (Baltimore) 1982;61:317-29.  Back to cited text no. 14
    
15.
Wiltshaw E. The natural history of extramedullary plasmacytoma and its relation to solitary myeloma of bone and myelomatosis. Medicine (Baltimore) 1976;55:217-38.  Back to cited text no. 15
    
16.
Moshref M, Mashhadi-Abbass F, Sargolzaei S, Nafarzadeh S. Extramedullary plasmacytoma of the gingiva. Arch Iran Med 2007;10:91-3.  Back to cited text no. 16
    
17.
Gholizadeh N, Mehdipour M, Rohani B, Esmaeili V. Extramedullary plasmacytoma of the oral cavity in a young man: A case report. J Dent (Shiraz) 2016;17:155-8.  Back to cited text no. 17
    
18.
Kapadia SB, Desai U, Cheng VS. Extramedullary plasmacytoma of the head and neck. A clinicopathologic study of 20 cases. Medicine (Baltimore) 1982;61:317-29.  Back to cited text no. 18
    
19.
Wanebo H, Geller W, Gerold F. Extramedullary plasmacytoma of upper respiratory tract. Recurrence after latency of thirty-six years. N Y State J Med 1966;66:1110-3.  Back to cited text no. 19
    


    Figures

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



 

 
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