International Journal of Surgery Case Reports 115 (2024) 109276

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International Journal of Surgery Case Reports

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Case report

Solitary bone plasmacytoma mimicking a lesion of odontogenic origin: A

case report
Zhiguo Liu a, b, Minyi Cui a, b, Siyi Zheng a, b, Hongyan Liu a, b, *
a
Hospital of Stomatology, Sun Yat-sen University, Guangzhou 510055, Guangdong, China
b
Guangdong Provincial Clinical Research Center of Oral Disease, Guangzhou 510055, Guangdong, China

A R T I C L E I N F O A B S T R A C T

Keywords: Introduction and importance: Solitary bone plasmacytoma (SBP) is an early-stage plasma cell malignancy. It is an
Solitary bone plasmacytoma extremely rare condition and its diagnosis may not be straightforward. This report presents a case of maxillary
Periapical lesion SBP.
Maxillary
Case presentation: A 48-year old man sought care for persistent swelling and pain in the periapical region of the
Misdiagnosis
left maxillary molars. He had been diagnosed with “apical periodontitis” and root canal treatment of teeth #26
Case report
and #27 was ineffective. Extra-oral examination revealed swelling at the left maxilla. Intraoral examination
revealed a hard, non-fluctuant swollen region in the buccal alveolar mucosa adjacent to the apices of teeth
#25–27. Cone-beam computed tomography revealed extensive bone destruction in the left maxilla. The patient
underwent partial maxillary resection and radical maxillary sinusotomy. Further testing (positron emission CT
scan, histopathological and immunohistochemical examination) confirmed the diagnosis of SBP. The patient had
a recurrence two years later, which was managed with left subtotal maxillectomy and radiotherapy. There was
no evidence of recurrence during 20 months of follow-up.
Discussion: SBP may mimic an odontogenic lesion when found in the jaw bone. To confirm the diagnosis, routine
blood test, complete body skeletal survey, metastatic investigations and histopathology should be performed.
Radiotherapy is the primary treatment.
Conclusion: SBP may occur in the maxilla mimicking an odontogenic lesion. Surgery may be part of the diagnostic
procedure and an adjunct to definitive radiation. Radiotherapy is the primary treatment.

1. Introduction report presents a rare case of maxillary SBP mimicking a lesion of

Plasma cell neoplasms are abnormal proliferations of monoclonal B-
cells that rarely occur in the jaw bones. These neoplasms have various
clinical presentations, including multiple myeloma (generally located in
the bone marrow) and solitary plasmacytoma (SP, characterized by a
single mass of clonal plasma cells, with minimal or no bone marrow
plasmacytosis or systemic involvement). SP can present as solitary bone
plasmacytoma (SBP, limited to one site in the axial skeleton) or extra­
medullary plasmacytoma (EMP, usually limited to soft tissues) [1,2].
Most SBP patients develop myeloma (median time to progression, 2–4
years) and the overall median survival ranges from 7.5 to 12 years [3].
SBP mostly affects red marrow-containing bones including the femurs,
pelvis, ribs, and vertebrae. It is rare in oralfacial region and only 4.4 % of
SBP have been reported in mandible [4,5]. Maxillary SBP is an
extremely rare condition and its diagnosis may be difficult [6]. This
odontogenic origin. The diagnosis and management of the case are
described as below.
2. Case presentation
This work has been reported in line with the SCARE Guidelines [7]
and the PROCESS criteria [8].
A 48-year-old man visited the Department of Operative Dentistry and
Endodontics in our hospital with persistent pain and swelling in the
periapical region of the left upper molars for two months. He was
diagnosed with “apical periodontitis” of teeth #26 and #27 at a pe­
ripheral dental clinic and endodontic treatment was started. However,
the pain and swelling did not subside. The patient denied any further
history of surgical treatment, sustained fever, systemic diseases, al­
lergies, or unintended weight loss. The patient had no significant past

* Corresponding author at: Hospital of Stomatology, Sun Yat-sen University, Guangzhou 510055, Guangdong, China.
E-mail address: liuhyan@mail.sysu.edu.cn (H. Liu).

https://doi.org/10.1016/j.ijscr.2024.109276
Received 13 December 2023; Received in revised form 11 January 2024; Accepted 12 January 2024
Available online 22 January 2024
2210-2612/© 2024 The Authors. Published by Elsevier Ltd on behalf of IJS Publishing Group Ltd. This is an open access article under the CC BY-NC-ND license
(http://creativecommons.org/licenses/by-nc-nd/4.0/).
Z. Liu et al.
Fig. 1. Periapical radiograph revealed incomplete endodontic treatment of
teeth #26 and #27 and presence of lower radiodensity area at the apices of
teeth #26 and #27.
medical history.
Extraoral examination revealed the presence of a hard bony swelling
measuring 3.0 × 4.0 × 2.0 cm at the left maxillary region with a well-
defined tender boundary and obliteration of the buccal sulcus. No
numbness was observed in the left infraorbital region. There were no
palpable lymph nodes, blockage of the left nasal cavity, or numbness.
Intraoral examination revealed a hard swelling in the region of the
buccal alveolar mucosa associated with teeth #25–27. The access cav­
ities of teeth #26–27 were previously sealed with temporary cement and
were negative to a cold and electrical pulp test. The periapical area of
teeth #26–27 was slightly sensitive to percussion and showed no
mobility. The teeth #25 and #28 were responsive to cold stimuli
without any tenderness. Periapical radiograph examination revealed
incomplete endodontic treatment of teeth #26 and #27 and the pres­
ence of a lower radiodensity area at the apices of teeth #26 and #27
(Fig. 1). Cone-beam computed tomography (CBCT) machine (DCT-pro,
VATECH, Korea), which demonstrated poorly defined destruction
(approximating 29.5 × 28.5 × 20.5 mm), with intra-tumoral thin and
straight septa at the apices of teeth #25–27 in the left maxilla. The
associated buccal and palatal cortexes were destroyed without root
resorption. Soft tissues emanating from the bone was observed and a
strip-like high density was seen in the soft tissue mass, suggesting
Fig. 2. Initial Cone Beam Computed Tomographic imaging revealed a poorly defined destruction at the apices of the teeth #25–27 in the left maxilla. The adjacent
buccal and palatal cortex was destructed. Soft tissue emanating from the bone was demonstrated, and a strip-like shadow with high density was also found in the soft
tissue mass. The tumor invaded to the left maxillary sinus (arrow). The CBCT showed no signs of root resorption (A) Coronal section, (B) sagittal section, (C)
axial section.
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International Journal of Surgery Case Reports 115 (2024) 109276
invasion of the left maxillary sinus by the tumor. The CBCT images
showed no signs of root resorption (Fig. 2). Biochemistry analysis of the
blood and urine was unremarkable.
The patient was directed to the Oral and Maxillofacial Surgery
Department. Histopathological analysis of frozen biopsy samples
showed a round and sheet-like arrangement of tumor cells while a few
cells had large nuclei with atypia and rare mitosis, suggesting a possible
diagnosis of odontogenic tumors. Partial maxillary resection and radical
maxillary sinusotomy were performed. Pathological analysis showed a
diffuse pattern of plasmacytoid cells characterized by large, atypical,
and eccentric nuclei, dichromatic cytoplasm, and occasional mitosis in
bone tissues (Fig. 3A). Immunohistochemical examination confirmed a
population of cells positive for kappa (Fig. 3B) and negative for lambda
immunoglobulin light chain (Fig. 3C). The above findings were consis­
tent with plasmacytoma. A positron emission CT scan was performed
and proved no tumor cell infiltration to other tissues. Bone marrow bi­
opsy examination showed no abnormalities. Based on the clinical and
histologic findings, the patient was diagnosed with solitary bone plas­
macytoma. Radiotherapy was suggested, however, the patient refused.
Two years later, the patient was re-evaluated with tumor recurrence.
CBCT showed the extraction of teeth #25–28 and defective alveolar
crests on these teeth and the inferior wall of the left maxillary sinus. The
alveolar bone of the first premolar was irregularly absorbed. A soft tissue
mass was observed in the left maxillary sinus. The lesions penetrated
into the left nasal cavity and the bone on the inner wall of the left
maxillary sinus was absorbed (Fig. 4). Left subtotal resection of the
maxilla was performed. Pathological analysis showed findings similar to
those in previous reports. The patient was referred to the Cancer Center
for radiotherapy to a dose of 50Gy in 25 fractions. During the 20-month
follow-up period, the patient reported no discomfort or pain.
3. Discussion
SBP is an early-stage plasma cell-localized malignant lesion that may
mimic an odontogenic lesion when occurs in the jaw bone. In the present
study, the lesion was located around the apical area of the maxillary
molars, mimicking periapical radiolucency. Further workup including
CBCT and histopathological testing was used to confirm the diagnosis
and for treatment planning. Non-odontogenic lesions in the dento-
alveolar region are likely to be misdiagnosed as odontogenic origins.
Sirotheau et al. evaluated 56 cases referred to oral pathology initially
misdiagnosed as periapical lesions. They found that 29 % (16/56) of
cases represented malignant lesions. The most common malignancies
included metastatic injuries (31.5 %) and carcinomas (25 %) and pre­
sented with pain and swelling [9].
Z. Liu et al. International Journal of Surgery Case Reports 115 (2024) 109276

Fig. 3. Histological examination of the bone tissue. (A) H&E-stained section shows a diffused pattern of plasmacytoid cells characterized by large, atypical and
eccentric nuclei, dichromatic cytoplasm and occasional mitosis in bone tissues (100×). Immnunohistochemical stain revealed a population of cells positive for kappa
(B) and negative for lambda immunoglobulin light chain (C) (100×).

There are very few retrospective studies reporting the occurrence of
SBP in jaw bones. In the maxillofacial region, SBP is relatively common
in the mandible, especially in bone marrow-rich areas including the
retromolar trigone, angle, and ramus [10]. Swelling and pain are the
primary clinical presentations of SBP in the jaw, occasionally with
mobility of the associated teeth and numbness of the lips [11]. The In­
ternational Myeloma Working Group recommends the following
guidelines to diagnose SBP: normal bone marrow without evidence of
clonal plasma cells, a biopsy-proven bone solitary lesion with evidence
of clonal plasma cells, magnetic resonance imaging, or CT of the spine
and pelvis showing normal skeletal characteristics (except for the pri­
mary solitary lesion), and the absence of end-organ damage such as
hypercalcemia, renal insufficiency, anemia, or bone lesions [12]. The
findings from clinical examination in this case were consistent with the

3
Z. Liu et al.
Fig. 4. Cone Beam Computed Tomographic imaging of two years after the first operation. The CBCT revealed the defective alveolar crest of these teeth and inferior
wall of the left maxillary sinus. The crest bone of alveolar of the teeth #24 was absorbed irregularly. Soft tissues were penetrated into the left nasal cavity, and bone
of the inner wall of the left maxillary sinus was resorbed (arrow). (A) Coronal section, (B) sagittal section, (C) axial section.
above criteria.
SBP is easy to be misdiagnosed as odontogenic tumor. The radio­
graphic presentation was close to that of odontogenic myxoma in the
CBCT images: fine and straight septa separated the tumor into trian­
gular, square, or rectangular spaces, mimicking a “tennis racket” or
“honeycomb” pattern [13]. Therefore, SBP should also be differentiated
from odontogenic myxoma radiographically.
SBP itself is a radiosensitive lesion, so radiotherapy is always
considered the primary treatment [14,15]. Local control rates can be
achieved in 80–90 % of cases with radiation therapy alone [16]. But
Ghiassi-Nejad et al. reported that in the United States, only 66.5 % of
patients with solitary plasmacytoma received radiotherapy [17].
Sometimes, surgical intervention is indicated, especially in cases of
compromised neurological and structural instability [18]. In a system­
atic survey about solitary plasmacytoma of the jaws, nearly 58 % of
patients underwent surgical treatment, including only surgical treat­
ment or associated radiotherapy and/or chemotherapy [15]. In many
instances, patients have had surgery as part of the diagnostic procedure
and an adjunct to definitive radiation. In the present case, the patient
underwent surgery without subsequence radiotherapy initially and
suffered a relapse of the SBP. Radiotherapy was performed for the
recurrent lesion.
4. Conclusion
In conclusion, SBP may occur in the maxillary bones mimicking an
odontogenic lesion without specific clinical manifestations. Non-
endodontic lesions should be considered during differential diagnosis
of apical periodontitis. Microscopic analysis of biopsy samples should be
performed to rule out the possibility of malignancy when necessary.
Radiotherapy can be considered the primary treatment after definite
diagnosis.
Consent
Written informed consent was obtained from the patient for publi­
cation of this case report and accompanying images. A copy of the
written consent is available for review by the Editor-in-Chief of this
journal on request.
Ethical approval
The protocols were approved by the University Ethic Committee at
the Sun Yat-sen University, China (KQEC-2020-08).
4
International Journal of Surgery Case Reports 115 (2024) 109276
Funding
The authors state that this study has received no financial support.
Author contribution
Zhiguo Liu: Investigation, Writing- original draft.
Minyi Cui: Investigation, Resources, Writing- original draft
Siyi Zheng: Writing- original draft.
Hongyan Liu: Investigation, Resources, Writing-review and editing,
Validation.
Guarantor
Hongyan Liu.
Research registration number
Not applicable.
Conflict of interest statement
The authors declare that they have no any financial and personal
relationships with other people or organisations that could inappropri­
ately influence (bias) their work.
Data availability
The datasets used and/or analyzed during the current study are
available from the corresponding author on reasonable request.
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