Solitary Plasmacytoma of the Parotid Gland with Crystalline Inclusions: A Case Report
© Kanthan and Torkian; licensee BioMed Central Ltd. 2003
Received: 14 May 2003
Accepted: 01 August 2003
Published: 01 August 2003
Solitary extramedullary plasmacytoma of the parotid gland is a rare condition. Intracytoplasmic Crystalline inclusions in the tumor are even rarer and have been reported only once in the parotid gland.
We report here, a case of plasmacytoma of the parotid gland with intracellular crystalline inclusions in a 73-year-old woman
Solitary extramedullary plasmacytoma of the parotid gland and crystalline inclusions in the tumor is of rare occurrence. The importance of such a finding with relation to tumor progression, clinical course of the disease or prognosis in general remains to be understood.
KeywordsParotid neoplasm plasmocytoma inclusion salivary gland.
Review-published Cases of Parotid Plasmacytomas
1-Ustun MO et al. 
2-Hari CK, Roblin DG. 
3-Gonzalez-Garcia J et al. 
4-El-Naggar AK 
5-Kerr PD, Dort JC 
6-Rothfield RE et al. 
7-Simi U et al. 
8-Scholl P, Jafek BW 
9-Ebbers J 
10-Kanoh et al. 
11-Edney JA et al 
12-Ferlito A et al 
13-Pahor AL. 
14-Vainio-Mattila J 
Solitary primary extramedullary plasmacytomas are rare and found principally in elderly people. They occur predominantly in the head and neck area with a tendency to involve the submucosal tissues of the upper airway accounting for 0.5% of all upper respiratory tract neoplasms. In two third of the cases, these neoplasms are located in the upper respiratory tract or oral cavity primarily in the nose, sinuses and nasopharynx .
Histopathological analysis alone is not sufficient in order to make a diagnosis of primary extramedullary plasmacytoma as multiple myeloma must be excluded by a through skeletal and marrow workup, immuno-electrophoresis and quantitative immunoglobulins with absent urinary Bence Jones proteins. Fine needle aspiration cytology of plasmacytoma of the parotid gland is often reported unremarkable as in our case.
Treatment of solitary extramedullary plasmacytoma consists primarily of eradication of the local lesion and in this context surgery seems to be the primary line of treatment. Radiation therapy either alone in cases deemed unsuitable for surgery or as an adjunct with surgical removal has also been used. Local recurrences are also usually treated by radiation therapy  Although not proven, chemotherapy is theoretically advantageous, both in enhancing local control and in potentially eradicating sub-clinical disease to delay or prevent the development of myeloma. The patterns of relapse of primary extramedullary plasmacytoma include transformation to multiple myeloma, metastasis and local recurrence. Recurrences are usually local and respond well to radiation therapy. Combined chemotherapy and radiation therapy is recommended in high-risk patients to increase the local control rate and cure rate  In our case radiotherapy could have been added after surgery in order to eradicate residual tumor and to prevent local relapse. However, the validity of such adjuvant therapy with complete excision of the tumor with clear margins remains contradictory. Radiotherapy was administered in our case, upon local recurrence. The second local recurrence was managed by chemotherapy.
Crystalline inclusions composed of monoclonal immunoglobulins have been reported in multiple myeloma and lymphoproliferative disorders . A single publication of these crystalline inclusions in primary extramedullary plasmacytoma of the parotid gland has been reported by el-Naggar et al.  The crystals may be rectangular or square in shape, finely uniform or globular. Crystals can be stained with anti light and/or anti heavy chains of globulin, which often manifest a light chain restriction. They can be found in histiocytes, which absorb the immunoglobulins . The crystalline inclusions in our case were positive for IGg with Kappa restriction, confirming the intracellular crystallization of the immunoglobulin. Electron micrographs of the crystalline deposits show the polygonal, rectangular or diamond shape of this finding in solitary plasmacytoma. The importance of such a finding with relation to tumor progression, clinical course of the disease or prognosis in general remains uncertain.
Primary solitary extramedullary plasmacytoma of the parotid gland is of rare occurrence. Accurate diagnosis by fine needle aspiration requires a high index of suspicion. The presence of intracytoplasmic crystalline inclusions can further add confusion to the accurate pathological diagnosis of this entity especially at frozen section analysis.
Conflict of interest
RK is the surgical pathologist who diagnosed and followed-up this case and BT participated actively in the production of this manuscript.
The authors would like to thank Mr Todd Reichert and Michelle Hesson for their expert technical assistance in the production of the illustrations. Written consent was obtained from the patient for the use of all clinical material for research and publication.
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