Case reports Annals and Essences of Dentistry

10.5368/aedj.2017.9.2.2.1
A CASE REPORT ON INTRAORAL PLASMA CELL GRANULOMA IN AN
UNUSUAL SITE
1 1
Venkata Ramanand Oruganti Senior Lecturer
2 2
Srinivas Munisekar Manay Lecturer
3 3
Juliana Beryl Paul Postgraduate Student
4 4
Thanuja Seethapathy Postgraduate Student

1,3,4
Department of Oral and Maxillofacial Pathology, SVS Institute of Dental Sciences, Mahabubnagar, Telangana, India.
2
Department of Oral Pathology, College of Dentistry, Aljouf University, Sakaka, Kingdom of Saudi Arabia.

ABSTRACT: Plasma cell granuloma is a rare non-neoplastic lesion that occurs most often in the lungs, but it is not
commonly seen in the maxillofacial region. Its etiology, biological behavior, ideal treatment and prognosis are still unclear
and rather controversial. Very few cases of intraoral plasma cell granulomas have been reported in the past. Hence, we
present an unusual case of plasma cell granuloma in a 60year-old female which was presented clinically as ill-defined
swelling over left upper buccal vestibule region extending to the midpalate. Histological examination revealed fibro-cellular
connective tissue stroma with inflammatory cell infiltrate containing plasma cells and lymphocytes. The plasma cells are
abundant varying in size and shape, with very few large and binucleated plasma cells. Both clinically and
histopathologically, it may be misinterpreted as various pathological entities thus proper evaluation of patient and
histopathological examination of the tissue to rule out other lesions is mandatory.
.

KEYWORDS: Plasma cell granuloma, immunohistochemistry, reactive lesion.

INTRODUCTION

Plasma cell granuloma (PCG) was first described by
Bahadori and Liebow in 1973. Intraoral PCGs have been
reported on the tongue, lip, and buccal mucosa and in the
gingiva. PCG in the oral cavity can be easily mistaken
clinically as peripheral giant cell granuloma, pyogenic
granuloma, thus necessitating the histopathological
examination to confirm the diagnosis. Hence, the clinical,
radiological, histopathological findings must be correlated
for an accurate diagnosis. This present case report
describes a rare case of plasma cell granuloma on the
buccal vestibule region extending to the palate which was
1
clinically thought as ameloblastoma.
Case Report
A 60-year-old female patient reported to the
department of Oral Medicine and Radiology, SVS institute
of Dental Sciences, Mahabubnagar, with a chief complaint
of pain and swelling in the upper left back tooth region for
the past 6 months. The swelling was initially small in size
which gradually increased in size. Extraorally, a 5X4 cm
solitary diffuse swelling was noticed in the left middle third
of the face. The swelling extended anteroposteriorly from
the ala of the nose to 5cms in front of the ear.
Superoinferiorly, it extended 2cms below the left eyelid to
1cm above the corner of the mouth. The skin over the
swelling was normal. On intraoral examination, a solitary
growth was extending from 26 to 28 over the left side of
upper buccal vestibule region which extended up to the
midpalate (Fig.1). The swelling was well-defined, oval in
shape measuring about 5X4cms approximately. The
colour of the swelling was slightly red as compared to that
of the surrounding mucosa. On palpation, the inspectory
findings were confirmed. The swelling was fluctuant in the
palatal region, soft in consistency in the center and hard at
the periphery and it was tender.
On radiographic investigations OPG revealed ill-
defined radiopacity in the left maxillary sinus causing
downward displacement of the arch and teeth (23 to 26)
and loss of lamina dura of involved teeth and root apices
(Fig .2). The occlusal radiograph revealed a well-defined
multilocular radiolucency distal to 27. It also showed
expansion of the buccal cortex and erosion of palatal
bone. CT scan revealed extension of the swelling from
maxillary arch posteriorly and involving and obliberating
maxillary sinus and superiorly extending till inferior border
of mandible. A provisional diagnosis of ameloblastoma
was made and the patient was sent to Department of Oral
and Maxillofacial Surgery. Routine blood investigation was
done. The findings were within normal limits.
An incisional biopsy was done under local
anaesthesia and the specimen was sent for

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Case reports Annals and Essences of Dentistry

Fig.1. Solitary growth from the left side of upper buccal vestibule region extending to the midpalate

Fig. 2. OPG revealed ill-defined radiopacity in the left maxillary sinus

Fig.3.High power magnification (40x) revealed a Fig.4.Immunohistochemical study reveals the

fibro-cellular connective tissue stroma with plasma cell infiltrate uniformly positive for
intense chronic inflammatory cell infiltrate chiefly CD138 antibody, a marker for plasma cells.
composed of plasma cells and lymphocytes. The
plasma cells with varying in size and shape and
few large and binucleated plasma cells

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Case reports
histopathological examination. Histopathological
examination revealed a fibro-cellular connective tissue
stroma with intense chronic inflammatory cell infiltrate
chiefly composed of plasma cells and lymphocytes. The
plasma cells were abundant varying in size and shape,
with very few large and binucleated plasma cells (Fig .3).
The connective tissue also shows numerous endothelial
lined budding capillaries surrounded by clusters of plasma
cells. Areas of hemorrhage, foci of ossification and
extravasated RBCs were also evident.
Immunohistochemical study of the biopsy showed the
plasma cell infiltrate uniformly positive for CD138, a
marker for plasma cells (Fig. 4).
A diagnostic work up was done to rule out multiple
myeloma and extra medullary plasmacytoma. Chest x-ray
and ECG revealed no significant features. The urine
analysis for Bence-Jones proteins revealed negative thus
ruling out multiple myeloma. Based on the clinical,
histopathological findings the final diagnosis of Plasma cell
granuloma was given.
Discussion
Plasma cells are terminally differentiated B
lymphocytes that provide protective immunity through
continuous secretion of antibodies. They are commonly
seen in chronic inflammatory infiltrates and were first
described by Zoon in balantis plasma cellularism in the
year 1952. Since then, plasma cell infiltrates have been
documented in the vulva, buccal mucosa, nasal aperture,
gingiva, lips, tongue, epiglottis, larynx and other orificial
surfaces. The plasma cell granulomas were considered to
be highly uncommon, non- neoplastic, reactive lesion,
which were first brought to the attention of health care
practitioners during the late 1960s and early 1970.
Bhaskar, Levin and Firch first reported this pathological
entity on the gingival tissue and only very few case reports
have been documented since then. Avecdo and Buchler,
Mark and Steven, Karthikeyan and Pradeep and
Baltaciaglu et al. have reported this lesion on the gingiva.
However, the present case was seen in the posterior
palatal region which was well-defined, oval in shape
present on the left upper buccal vestibule region and
1,2,3,4,5
extended up to the midpalate.
The pathogenesis of the PCG remains unclear. The
large number of plasma cells may represent an
autoimmune reaction or an alteration of blood flow
imposing congestive vasodilatation. Lesions occurring due
6
to parasitic infiltration can also not be ruled out. The
presence of polyclonal plasma cells, lymphocytes and
7
histiocytes suggests an infectious and autoimmune origin.
PCG is thought to result from inflammation following minor
trauma or surgery or to be associated with
8,9,10
malignancy, however, this was not so in the present
case and our patient was free of any history of trauma or
malignancy. However, her oral hygiene was extremely
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Annals and Essences of Dentistry
poor with generalized chronic destructive periodontitis,
which could have contributed to the genesis of this lesion.
Clinically, gingival PCG presents as a mass, which
is nodular, polypoidal lesion with a smooth surface and
well circumscribed. It does not produce significant
11
systemic symptoms. Similar findings were seen in the
present case.
PCG is microscopically characterized by vascular
stroma with reactive inflammatory cells, including plasma
cells predominantly and usually surrounded connective
tissue septa. No cytological abnormalities are usually
present, Russell bodies, which are intra-cytoplasmic
12
eosinophilic hyaline droplets, may also be seen. In our
case, microscopic examination revealed fibro-cellular
connective tissue stroma with intense chronic
inflammatory cell infiltrate chiefly composed of plasma
cells and lymphocytes. The plasma cells were abundant
varying in size and shape, with very few large and
binucleated plasma cells. The connective tissue also
shows numerous endothelial lined budding capillaries
surrounded by clusters of plasma cells. Areas of
hemorrhage, foci of ossification and extravasated RBCs
are also evident.
PCG is a diagnosis of exclusion, distinguished
primarily on the histological finding of a marked
13
submucosal plasma cell infiltrate. Histologically, it is
important to differentiate various plasma cell lesions such
PCG, plasma cell gingivitis, and extramedullary
plasmacytoma (a possible precursors of multiple
14
myeloma).
Extramedullary plasmacytoma presents as a soft
tissue mass outside of bone. The main differentiation
between reactive PCG and extramedullary plasmacytoma
16
is based on whether lesion is polyclonal or monoclonal.
Histologically, extramedullary plasmacytoma consists of
mixture of typical and atypical plasma cells while plasma
cell granuloma consists of normal plasma cells and small
lymphocytes that are surrounded by connective tissue
septa. The immunohistochemical analysis have shown
that in the case of malignancy ratio of the kappa to lambda
light chain may be greater than 10:1 or 1:10, whereas in a
17
reactive lesion the ratio is 2:1.
Some consider PCG to be a subtype of plasma cell
16
gingivitis, plasma cell gingivitis is a polyclonal lesion of
gingival tissue that is usually not a localized nodular
lesion, as seen in PCG but presents as generalized
4
edematous and erythematous elevations. In the present
case a single, localized nodular mass was present.
Plasma cell gingivitis is considered as an allergic
hypersensitivity reaction to various flavoring agents used
18
in chewing gums and dentifrices. But in our case, there
was no identifiable inciting agent.
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Case reports Annals and Essences of Dentistry
PCG is usually treated by simple excision and 13. Anila Namboodripad PC, Jaganath M, Sunitha B,
19
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and recurrences may complicate the outcome of the Gingival plasma cell granuloma. Dent Res J (Isfahan)
20
disease. 2012; 9:816-20.
15. Renfrow JJ, Mitchell JW, Goodman M, Mellen LA,
CONCLUSION Wilson JA, Mott RT, et al. Relapsing intracranial
plasma cell granuloma: A case report. Oncol Lett
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exact etiology, behavior and prognosis is still 16. Peacock ME, Hokett SD, Hellstein JW, Herold RW,
controversial. Attributing to the fact that it can masquerade Matz enbacher SA, Scales DK, et al. Gingival plasma
as various pathological entity, it is difficult to establish the cell granuloma. J Periodontol 2001; 72:1287-90.
exact diagnosis alone on the basis of clinical or 17. Vishnudas B, Sameer Z, Shriram B, Rekha K.
histopathological examination. Therefore, it is necessary Amlodipine induced plasma cell granuloma of the
to perform complete lab investigations differentially to gingiva: A novel case report. Journal of natural
diagnose it from other lesions that have a poor prognosis science, biology, and medicine. 2014 Jul;5(2):472.
and to avoid unnecessarily extensive and potentially 18. Silverman S Jr, Lozada F. An epilogue to plasma-cell
destructive surgery. gingivostomatitis (allergic gingivostamtitis). Oral Surg
Oral Med Oral Pathol 1977; 43:211-7.
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