CASE REPORT/CLINICAL TECHNIQUES

Katherine A. Divine, DDS, MS

Multilocular Myospherulosis of
the Mandible: A Case Report

ABSTRACT
SIGNIFICANCE
Myospherulosis in the jaw is a rare, foreign body reaction that typically follows tooth extraction
and the use of petrolatum-based medicaments. It appears as a radiolucency and can be This is the first case of
mistaken for endodontic pathosis or other lesions without thorough clinical and radiographic myospherulosis in the
evaluation as well as biopsy submission. Treatment is surgical removal and the recurrence is endodontic literature. The
rare. This case report presents a unique finding of a posterior mandible multilocular lesion presented as an apical
radiolucency appearing as an apical lesion on 2-dimensional panoramic imaging until radiolucency of tooth 18.
visualized with 3-dimensional imaging. Clinical testing and patient referral led to a biopsy and Thorough clinical and
diagnosis of myospherulosis. This is the first case report of myospherulosis presented in the radiographic evaluation, as
endodontic literature. (J Endod 2022;-:1–4.) well as biopsy submission,
identified the lesion, and
KEY WORDS unnecessary endodontic
Case report; diagnosis; myospherulosis; pathology therapy was avoided.

Myospherulosis, also known as spherulosis or spherulocytosis, was first reported by McClatchie et al in

19691. They identified atypical lesions in the arm and leg skeletal muscles from East Africa in locations of
previous injections from various necessary surgeries. The condition was termed “myospherulosis”
because of its location in muscle and histologic features of “spherules” appearing in cyst-like spaces2.
The lesion, albeit rare, has since been identified most commonly in the paranasal sinuses, nose, middle
ear3, and jaw2,4–6.
The presence of the lesion in head and neck areas has been identified as an iatrogenic disease
caused by a foreign body reaction to lipid components within applied medicaments leading to the
alteration of erythrocytes7. Petrolatum-based antibiotic ointments are the most frequently cited etiologic
agents within the literature3,6–8. The ointment most commonly identified is Terra-Cortril (oxytetracycline
and hydrocortisone) (Pfizer U.S. Pharmaceuticals Group, New York, NY)6. The antibiotics alone are
noncontributing to myospherulosis; it is the petrolatum or the combination of petrolatum and
other substances that induces the foreign body reaction4. Terra-Cortril has been shown to reduce
the incidence of dry sockets in mandibular third molar extraction sites, thus stimulating its use in the
1980s8–10. Petrolatum-based antibiotic ointments, including Terra-Cortril, are one ingredient within the
Sargenti paste endodontic technique4. Bone wax is another material containing petrolatum with historical
use in oral and endodontic surgery at risk of inducing myospherulosis11. The use of these materials is not
recommended12–15.
The histopathologic findings revealed a mixed inflammatory infiltrate, cyst-like spaces, and
numerous erythrocytes exhibiting a variety of morphologic alterations. Clinical appearance reported
during surgical access and biopsy is a brownish-black substance that is tar-like in texture and often
associated with a membranous mucoid substance6. Treatment of myospherulosis includes complete
and conservative curettage with specimen submission for histopathologic examination. Recurrence is
rare2. From Private Practice, Boise, Idaho
Within published research, reports of this lesion are rare. Since first described in 1969,
The authors deny any conflicts of interest.
myospherulosis has been found in muscle, subcutaneous tissue, sinuses, intracranial, breast, lip,
vagina, lacrimal caruncle, eyelid, retroperitoneal fat tissue, liver, and in the mandible16. There are only 15 Address requests for reprints to Dr
Katherine A. Divine, Private Practice, Blue
identifiable published cases of myospherulosis in the mandible at the time of this case report
Creek Endodontics, 3240 E. Louise Drive,
submission2,16. Eleven of the 15 have a known etiology of petrolatum-based antibiotics16. The #102, Meridian, ID 83642.
pathogenesis of myospherulosis lesions vary in length of time, symptoms, and are typically reported E-mail address: kdivine1@gmail.com
presenting in the jaw with a unilocular radiographic appearance6. This case is only the second 0099-2399/$ - see front matter
multilocular mandibular lesion specifically identified at the time of publication submission2,16. Overall, this Copyright © 2022 American Association
is the 16th reported case of myospherulosis in the mandible and the first case published in the of Endodontists.
endodontic literature. https://doi.org/10.1016/
j.joen.2022.04.006

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CASE REPORT symptomatic apical periodontitis. An occlusal as follows: “The decalcified specimen consists
adjustment was made on the distal-lingual of viable bone and fatty marrow. Multiple cystic
A 55-year-old White woman was seen in
cusp to relieve an excursive interference spaces containing variably-sized brown-
January 2022 for endodontic evaluation of the
identified with articulating paper, and the stained spherules representing altered
lower left second molar (tooth 18). Medical
patient reported improvement in her bite erythrocytes and partially surrounded by
history was noncontributory. She recently had
pressure sensitivity. She was advised to multinucleated giant cells are observed.
a crown placed on tooth 18 approximately 1
monitor for any development of temperature Pathologic Diagnosis: Viable bone, marrow
month prior and developed bite pressure
sensitivity and, if so, return for reevaluation. and altered erythrocytes compatible with
sensitivity afterward. She had third molar
Otherwise, bite pressure sensitivity was myospherulosis.” (Fig. 3).
extraction in her teenage years and endodontic
anticipated to resolve with the bite adjustment.
therapy of tooth 19 more than 10 years ago. A
In addition, a discussion occurred between the
panoramic radiograph from her referring
endodontist and patient regarding the atypical
DISCUSSION
dentist taken approximately 1 year before
finding on CBCT and that follow-up would be This case illustrates the value and importance
crown placement showed a periapical
necessary with an oral and maxillofacial of thorough clinical and radiographic
radiolucency on the distal root of tooth 18, and
radiologist for additional diagnostic evaluation for accurate diagnosis and proper
a periapical image (Carestream RVG 6100;
interpretation and treatment suggestions. treatment. If the panoramic radiograph was
Carestream Dental, Atlanta, GA) obtained at
The CBCT was referred to an oral and used solely for diagnosing apical pathosis,
the time of endodontic consult identified
maxillofacial radiologist for interpretation of the tooth 18 would have been unnecessarily
uniform periodontal ligament and intact lamina
atypical multilocular lesion in the buccal cortical treated without addressing the actual disease.
dura (Fig. 1). A limited-field-of-view cone-beam
bone adjacent to and posterior of tooth 18 Sensibility testing and multidimensional
computed tomography (CBCT) (CS 8100;
(Fig. 2). The oral radiologist interpretation radiographic imaging were essential for
Carestream Dental, Atlanta, GA) (Fig. 2) was
report findings were as follows: “There is well- complete evaluation. With CBCT, the
taken to evaluate the lower left at the time of
defined, multilocular radiolucency within the radiolucency was determined to be
consultation given the possibility of tooth
buccal cortex adjacent to tooth 18 and nonodontogenic. A lesion of nonodontogenic
fracture, previously endodontically treated
extending distally beyond the study. Internally origin once recognized should be referred for
tooth 19, and posterior mandible radiolucency
there are wispy separations. A linear further evaluation and subsequent treatment
from the referring office panoramic image17.
distolingual radiolucency extends from the as directed.
Clinical evaluation and testing revealed
primary radiolucency to the cancellous bone. The posterior mandible is a frequent
no swelling, no sinus tract, normal periodontal
The buccal cortex demonstrates mild area for radiolucent lesions to present and
probing depths, and no palpation sensitivity.
expansion and periosteal reaction with an myospherulosis is not typically included in the
Cold testing was completed with a #2 cotton
adjacent Codman triangle of the most mesial differential diagnoses because of its rarity. The
pellet and Endo Ice Spray (Coltene, Cuyahoga
radiolucency noted.” The radiologist differential differential diagnosis for multilocular
Falls, OH) with placement on the mid-buccal
diagnosis included intracortical osteosarcoma radiolucencies of the posterior mandible
surface of each tooth18. Tooth 18 was
or central giant cell granuloma. Biopsy was typically includes ameloblastoma, odontogenic
responsive and nonlingering to cold testing
recommended for a definitive diagnosis. myxoma, odontogenic keratocyst, and central
consistent with adjacent teeth. Percussion
The patient was referred to an oral and giant cell granuloma2. This case report, along
testing was completed with the mirror handle
maxillofacial surgeon who performed an with LeBlanc and Ghannoum’s2 2016 case
on the occlusal and buccal surfaces on the
excisional biopsy within days of receiving the report, demonstrate a need to include
lower left teeth. Tooth 18 was more sensitive to
radiology report. The tissue was submitted for myospherulosis in the differential diagnosis for
percussion testing than adjacent teeth. Tooth
histopathology to an oral and maxillofacial multilocular radiolucencies when adjacent to a
18 was diagnosed as normal pulp and
pathologist. The histopathology was reported site of previous tooth extraction.

FIGURE 1 – (A ) Panoramic radiographs. (B ) Cropped section from panoramic of tooth 18 with apical radiolucency. (C ) Periapical image of lower left, and tooth 18 appears without
apical radiolucency.

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FIGURE 2 – Cone-beam computed tomography screenshots depicting the well-defined, multilocular radiolucency in the buccal cortex. (A ) Appearance of Codman’s Triangle. (B and
C ) Multilocular radiolucency of the buccal cortex with expansion. (D ) Three- dimensional reconstructed view showing the multilocular lesion with cyst-like spaces. (Image layout
courtesy of TrueView Dental Radiology)

Research has identified specific
materials to be at higher risk of causing
foreign body reactions, such as the
aforementioned petrolatum-based antibiotic
ointment and bone wax12–14. The
American Association of Endodontists has
expressed opposition to the Sargenti
paste technique and other paraformaldehyde-
containing materials since 1991, and
continuously reaffirms this in their published
position paper14. Efforts should continue to
be made to avoid known caustic materials and
limit interactions of irritating materials outside
the apices of teeth as well as surrounding hard
and soft tissues12,15.
Although the direct etiology cannot be
confirmed for this case, it is possible the
relative time of the patient’s third molar
extractions would correspond with reported
Terra-Cortril usage in oral surgery9,10. The
location of this radiolucency in the buccal
cortical bone is different from the intraosseous
presentation of LeBlanc and Ghannoum’s2
multilocular myospherulosis case report. The
intraosseous location within the body of the
mandible for LeBlanc and Ghannoum’s2 case
seems to relate to the intrasocket placement of
Terra-Cortril following extraction. For this case,
the more superficial cortical bone location is
unique. The patient does not report any history
of topical medicament application in this site or
injections beyond dental anesthetics that could
have played an etiologic role.
Although the etiology will remain
unknown, the value of thorough diagnostic
testing and CBCT are demonstrated in this
case report. Furthermore, as the first published
case of myospherulosis in endodontic
literature, endodontists should now add this to
their differential diagnosis of radiolucencies
near sites of previous oral surgery.

CONCLUSION
The goal of this case report was to provide
education and awareness of myospherulosis for
differential diagnoses of multilocular posterior
radiolucencies and to illustrate the importance
of thorough clinical and radiographic evaluation
as well as biopsy submission for accurate
diagnosis and treatment.

ACKNOWLEDGMENTS
The author thanks True View Oral Radiology
(Boise, ID) for their CBCT interpretation service,
Whitewater Oral Surgery (Boise, ID) for surgical
case management, and Louisiana State
University Oral Pathology Biopsy Service for
FIGURE 3 – Brown spherules represent the altered erythrocytes characteristic of myospherulosis along with decalcified their review and diagnosis of the case. The
bone and a loose granulation tissue with focal compressed multinucleated giant cells (hematoxylin-eosin, original author denies any conflicts of interest related to
magnification !40). (Image courtesy of Louisiana State University Oral Pathology Biopsy Service) this study.

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and sealers. Available at: www.aae.org. Accessed January 15, 2022.
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