Vol. 117 No.

6 June 2014

Multiple simple bone cysts of the Jaws: Review of the literature

and report of three cases
Seo-Young An, DDS, MS,a,1 Jae-Seo Lee, DDS, PhD,b,1 Erika Benavides, DDS, PhD,c Alireza Aminlari, DDS, MS,d
Neville J. McDonald, DDS, MS,e Paul C. Edwards, MSc, DDS,f Min-Suk Heo, DDS, PhD,g
Hong-In Shin, DDS, PhD,h Jin-Woo Park, DDS, PhD,i Jae-Kwang Jung, DDS, MS,j Karp-Shik Choi, DDS, PhD,k
and Chang-Hyeon An, DDS, PhDl

Objectives. This study contributes three well-documented cases of multiple simple bone cysts (SBCs) of the jaws and reviews
previously published cases.
Study Design. A comprehensive literature search of multiple SBCs was conducted using the PubMed database. Synonyms of
SBC were used as search key words in combination with “mandible or jaw,” “bilateral, multiple, multifocal, atypical, and
unusual.”
Results. A total of 34 cases of multiple SBCs (including two asynchronous cases) were identified, including the three new
cases reported here. Multiple SBCs primarily occurred in the second decade (52.9%) and bilaterally in the posterior mandible.
Lesions demonstrated female predominance (1.8:1) and were frequently accompanied by bony expansion (44.1%) and a
multilocular radiolucent appearance (20.6 %). Recurrence was reported in three patients (mean age: 39.3 years old).
Conclusion. Knowledge of the clinical and radiographic features of multiple SBCs is important in the diagnosis and
management of this entity. (Oral Surg Oral Med Oral Pathol Oral Radiol 2014;117:e458-e469)

Financial support: This research was supported by Kyungpook First referred to as traumatic bone cysts by Lucas,1
National University Research Fund, 2012. the simple bone cyst (SBC) is a single bone cavity,
1
These authors contributed equally to this paper.
a
Assistant Professor, Department of Oral and Maxillofacial Radi-
generally fluid-filled, lacking epithelial lining.2 In
ology, School of Dentistry, Kyungpook National University, Daegu, 1947, Hansen et al. suggested that the term SBC should
Republic of Korea. be restricted to those cases presenting as an empty
cavity that might contain some fluid and/or small
b
Assistant Professor, Department of Oral and Maxillofacial Radi-
ology, School of Dentistry, Dental Science Institute, Chonnam amounts of soft tissue occasionally.3 Since it lacks
National University, Gwangju, Republic of Korea.
c
Clinical Assistant Professor, Department of Periodontics and Oral
epithelial lining, the SBC is not a true cyst. In addition
Medicine, School of Dentistry, University of Michigan, Ann Arbor, to the term simple bone cyst,4 a variety of other de-
MI, USA. scriptors have been used for this entity, including
traumatic bone cyst,3 hemorrhagic bone cyst,5 solitary
d
Adjunct Lecturer, Division of Endodontics, University of Michigan,
Ann Arbor, Michigan and Specialist Endodontic Private Practice, bone cyst,6 idiopathic bone cyst,7 extravasation
Farmington Hills, Michigan, USA.
e
Clinical Professor, Division Head and Graduate Program Director,
cyst,8 progressive bone cavity,9 and unicameral bone
Endodontics, University of Michigan, Ann Arbor, Michigan, USA. cyst.10
f
Professor, Department of Oral Pathology, Medicine and Radiology, While most SBCs present as solitary lesions, in one
Indiana University School of Dentistry, Indianapolis, Indiana, USA.
g
review of the literature,11 Eleven percent of SBCs
Professor, Department of Oral and Maxillofacial Radiology and presented as multifocal lesions, with the first case being
Dental Research Institute, School of Dentistry, Seoul National Uni-
versity, Seoul, Republic of Korea.
described by Hankey12 in 1947.
h
Associate Professor, Department of Oral Pathology, School In the present study, we systematically reviewed all
of Dentistry, Kyungpook National University, Daegu, Republic of previously recorded cases of multiple SBCs of the jaws
Korea. and described three additional cases of multiple SBCs
i
Assistant Professor, Department of Periodontology, School affecting the mandible that were evaluated by cone-
of Dentistry, Kyungpook National University, Daegu, Republic of
Korea.
beam computed tomography (CBCT).
j
Assistant Professor, Department of Oral Medicine, School
of Dentistry, Kyungpook National University, Daegu, Republic of MATERIAL AND METHODS
Korea. The present study was based on a literature search of
k
Professor, Department of Oral and Maxillofacial Radiology, School
articles published in English, conducted using PubMed,
of Dentistry, Kyungpook National University, Daegu, Republic of
Korea. a database created by the National Center for Biotech-
l
Associate Professor, Department of Oral and Maxillofacial Radi- nology Information (NCBI:http://www.ncbi.nlm.nih.
ology, School of Dentistry, Kyungpook National University, Daegu, gov/). The term “simple bone cyst” and its synonyms
Republic of Korea. (traumatic bone cyst, haemorrhagic/hemorrhagic bone
Received for publication Dec 9, 2013; returned for revision Feb 18, cyst, solitary bone cyst, idiopathic bone cyst/cavity,
2014; accepted for publication Mar 9, 2014.
Ó 2014 Elsevier Inc. All rights reserved. extravasation cyst, progressive bone cavity, and uni-
2212-4403/$ - see front matter cameral bone cyst) were searched in combination with
http://dx.doi.org/10.1016/j.oooo.2014.03.004 “maxilla or mandible or jaw” and “bilateral, multiple,

e458
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Volume 117, Number 6
multifocal, atypical, and unusual.” Multiple SBCs
associated with florid osseous dysplasia and cases with
solitary and large SBC misusing “bilateral” in their title
were excluded. The parameters that were examined and
collected are: Patient demographic, clinical, and radio-
graphic data, including age, gender, chief complaint at
presentation, history of trauma, vitality of the involved
teeth, contents of the cavity (soft tissue and fluid),
treatment, histology, outcomes of treatment, number of
lesions, location, radiographic findings (scalloped
margin, multilocular pattern and bony expansion) and
imaging modalities. Additionally, three new cases of
multiple SBCs with CBCT imaging are presented.
RESULTS
A PubMed search yielded 384 articles, of which 16
cases from 14 publications met the requirements for the
search. Fifteen additional cases were identified from the
references of these articles. Including the 3 new cases
described here, a total of 34 cases5-7,12-37 were analyzed
(Tables I and II and Figure 1). Two asynchronous
bilateral SBCs were included in this study.
An exact age was reported in 30 patients. Eighteen
(52.9%) of the 34 cases of multiple SBCs were found in
the second decade of life and eight cases (23.5 %)
occurred in patients greater than 30 years of age
(Table I and Figure 2). Among the 28 patients in which
gender was reported, a female predominance of 1.8:1
was noted (Figure 1). Twenty-two (64.7 %) asymp-
tomatic cases were detected after routine radiographic
examination. Swellings were noted in six patients
(17.6%). Only two patients had a documented history
of a traumatic event (a high-speed water-skiing spill and
a blow to the mandible). Vitality of involved teeth was
reported in 20 patients, with loss of vitality reported in
only one patient, representing one of our cases.
Fluid was aspirated in 7 of 11 patients (13 of 23
SBCs in this group). Of these 11 patients, fluid was also
identified at surgery in only one patient (representing 2
of 23 SBCs). Fluid was discovered in the cavity during
surgery in 8 of 23 patients (34.8%), while the cavities
were empty in 15 of 23 patients (65.2%).
Most multiple SBCs were treated surgically, with a
single case reporting spontaneous resolution of the
lesion after 4 years and 5 months. Multiple SBCs
recurred in the three oldest patients in this cohort (mean
age: 39.3 years of age) who had been managed by
incisional biopsy or surgical curettage.
The presence of two SBCs per patient was the most
common presentation, with seven patients presenting
with three SBCs. Most lesions were located in the
posterior mandible (Figures 3 and 4). Maxillary lesions
in combination with mandibular SBCs were noted
in two patients. Radiographically, scalloping was re-
ported in 21 patients (61.8%) and bony expansion was
CASE REPORT
An et al. e459
observed in 15 patients (44.1%). A multilocular radio-
graphic presentation was noted in seven patients
(20.6%), five of whom also presented with bony
expansion. Four of six multiple SBCs involving the
mandibular ramus area revealed both a multilocular
pattern and bony expansion. Multiple SBCs occurred
asynchronously in two patients with underlying condi-
tions, osteogenesis imperfecta and idiopathic thrombo-
cytopenic purpura respectively.
CASE REPORTS
Case 1
An 11-year-old girl was referred to the Department of Or-
thodontics, Kyungpook National University Dental Hospital
for orthodontic treatment. She was subsequently referred to
the Department of Oral and Maxillofacial Surgery for evalu-
ation of two incidentally noted unilocular radiolucencies
involving the left mandibular canine area and symphyseal
region (Figure 5). The left mandibular canine was mesially
impacted and the lamina dura of the adjacent teeth were intact.
Her medical history was unremarkable and she could not
recall any history of trauma to the mandible. Physical exam-
ination was likewise unremarkable, with no evidence of
swelling, asymmetry or lymphadenopathy. Intraoral exami-
nation disclosed no soft tissue abnormality in the area of the
anterior mandible except for an impacted left mandibular
canine. Several carious teeth were noted in the posterior areas.
The vitality of the anterior mandibular teeth was confirmed by
electric pulp testing.
CBCT images were acquired with a CB MercuRay (Hitachi
Medico, Tokyo, Japan) using a 10 cm field of view at 120
kVp and 15 mA. The presence of two separate and distinct
moderately defined unicystic lesions was confirmed. Buccal
expansion and slight thinning of the lingual cortex was
evident. The contents of the lesion were homogenously dense,
similar to or slightly less dense than the adjacent soft tissue
(Figure 6). The radiographic impression for both lesions was
SBC. The proposed treatment included surgical exploration
and incisional biopsy of both lesions with extraction of the left
mandibular canine to confirm the diagnosis and to promote
bone regeneration. Both lesions were approached surgically
under general anesthesia and were found to be empty, without
evidence of lining or fluid content. The lesions were curetted
in an attempt to obtain tissue for histopathologic analysis.
Despite several attempts, no soft tissue lining was found and
no tissue could be obtained for histopathologic examination.
A diagnosis of multiple SBCs was established surgically. The
postoperative course was uneventful. A follow-up panoramic
radiograph at 24 months shows adequate bony healing of both
lesions (Figure 7).
Case 2
A 17-year-old male patient visited a dental private clinic for
treatment of a carious tooth. Large unicystic radiolucencies
were noted bilaterally in the body of the mandible; a uniloc-
ular radiolucency in the left mandibular premolar area and a
scalloped unilocular radiolucency in the right mandibular
body area. Loss of the lamina dura of the adjacent teeth was
Table I. Characteristics of multiple SBCs. Results of a systematic review including our three cases

e460
ORAL AND MAXILLOFACIAL RADIOLOGY
Case Chief History Vitality Soft tissue Fluid in cavity
no. Year Author Age Sex complaints of trauma of teeth in cavity (aspiration/surgery) Treatment Histology Healing check

An et al.
1 1947 Hankey12 15 M RR
NS
NS/
Surgical intervention e 6 months
2 1954 Hutchinson15 23 NS RR
NS NS NS/NS NS NS NS
3 1963 Thoma16 15 F NS
NS NS NS/NS Surgical intervention SBC 15 months (Pano.)
4 1966 Szerlip14 15 F RR NS þ NS NS/NS Self healing e 4 year 5 months
5 1969 Grasso et al.17 35 F RR
þ þ Reddish-brown Surgical curettage Sclerotic bone 7 months
(Rt.)/
(autogenous venous
blood injection-Rt.)
6 1970 Morris et al.18 21 F RR NS þ
NS/Sanguineous Surgical intervention e NS
7 1971 Huebner and 11 F RR
þ
NS/Clear fluid Surgical opening e 4 months (Pano.)
Turlington19
8 1973 Stewart et al.20 17 M RR
NS
Light, reddish, Surgical curettage Lamellar bone, loose 6 months (Pano.)
watery/
erythrocytes
9 1974 Schofield13 20 F RR þ þ

/
Surgical curettage trabecula bone, connective 4-5 months (Pano.)
tissue (multinucleated cells
& histocytic stromal cells)
10 1975 Ruprecht and 17 M Pain NS þ
NS/
Surgical intervention e 7 months (Pano.)
Reid21
11 1978 Heimdahl22 16 M Swelling
þ
Serosanguineous/ Surgical curettage e 3 months
Serosanguineous
12 1978 Pogrel6 18 F RR
þ þ
/Serosanguineous Surgical intervention Bone, fibrous tissue 1 year
13 1979 Markus5 13 F RR NS NS NS NS/
Surgical intervention Lamellar bone, fibrous tissue 6 months (Pano.)
14 1979 Raibley et al.23 13 F RR
NS
Serosanguineous/NS Open and close Granulation tissue, 2 months (Pano.)
hemorrhage, cholesterol
clefts associated with
foreign body giant cells
15 1980 Kuroi24 36 F RR
þ79%
Serosanguineous/NS Surgical curettage e 1st: Recurrence, 3
months
2nd: 9 months
16 1981 Patrikiou et al.25 15 NS Pain NS þ NS NS/Serosanguineous Surgical intervention e 16 months (Pano.)
(Lt.)
17 1988 Forssell et al.26 NS NS NS NS NS þ NS/NS Open and close cancellous bone (osteoblast þ
rim), lamellar compact
bone (basophilia around
Haversian canals)
18 1991 Brannon and 21 F RR
NS þ NS/
Surgical curettage reactive bone, fibrous, and NS
Houston27 granulation tissue
19 1992 Saito et al.28 <30 NS RR NS þ NS NS/NS Surgical intervention NS þ
20 1992 Saito et al.28 >30 NS RR NS þ NS NS/NS Surgical intervention NS þ
21 1992 Saito et al.28 >30 NS RR NS þ NS NS/NS Surgical intervention NS þ
Prakash et al.29 þ

June 2014
22 1992 32 M Pain, NS NS/Serous Surgical intervention Bone fragments, chronic 4 months

OOOO
Swelling (gelfoam packing) inflammatory granulation
tissue
Table I. Continued

Volume 117, Number 6

OOOO
Case Chief History Vitality Soft tissue Fluid in cavity
no. Year Author Age Sex complaints of trauma of teeth in cavity (aspiration/surgery) Treatment Histology Healing check
23 1992 Fielding30 15 F RR e þ

/
Surgical curettage Dense cortical bone 1.5 years (PA)
24 1993 Jones and 23 F Pain,
NS NS NS/
Surgical intervention Bone trabeculae (rimmed by NS
Baughman7 Swelling osteoid and osteoblasts),
fibrous connective tissue
25 2002 Oda et al.31 12 M Swelling þ þ NS NS/NS Open and close Granulation tissue 8 months (Pano.)
26 2003 Kraut and Robin32 43 F NS NS NS NS Red (Rt.), Straw- Incisional biopsy Fragments of bone, 1st: recurrence,
colored (Lt.)/
granulaiton tissue, 18 months
osteoblastic rimming 2nd: 14 months
(Pano.)
27 2008 Mupparapu et al.33 39 F Pain, NS þ þ Serosanguineous/
Incisional biopsy bone trabeculae, fibrous 1st: recurrence, 2 year
Swelling connective tissue, rims of 2months
(Rt.) osteoid acute and chronic 2nd: NS
inflammatory cells
28 2010 Kuhmichel and 32 F RR
þ
NS/
Excisional biopsy NS 12 months (Pano.)
Bouloux34
29 2012 de Oliveira et al.35 22 M RR
NS

/NS Surgical curettage NS 2 years
30 2012 Martins-Filho 10 M NS
NS NS NS/Serous-bloody NS NS NS
et al.36
31 2012 Mathew et al.37 15 F Swelling
NS
NS/Clear, yellowish Surgical intervention NS 4 months (Pano.)
32 Our case 1 11 F RR
þ
NS/
Surgical curettage NS 2 years (Pano.)
33 Our case 2 17 M RR
þ
NS/
Surgical curettage NS NS
34 Our case 3 18 M RR
þ/

NS/
Surgical intervention Viable bone, dense 3 months (CBCT)
fibrovascular connective
tissue
RR, routine radiographic examination; NS, not stated; Pano., panoramic radiography; CBCT, cone-beam computed tomography; PA, periapical radiography; SBC, simple bone cysts, Rt., the right side; Lt., the
left side.

An et al. e461
CASE REPORT
ORAL AND MAXILLOFACIAL RADIOLOGY OOOO
e462 An et al. June 2014

Table II. Characteristics of multiple SBCs. Results of a systematic review, including our three cases
Case No. of
no. Year Author lesion Location (ADA system) Radiographic findings Image modality
12
1 1947 Hankey 3 #18-20, #25-26, #30-31 scalloping Lat. view
2 1954 Hutchinson15 2 #18-19, #28-30 scalloping, loss of lamina dura15 Lat. view
3 1963 Thoma16 2 Both Mn. Post. Areas NA Lat. view
4 1966 Szerlip14 2 #18-20, #30 scalloping Peri., Occ., Lat.
view
5 1969 Grasso et al.17 3 Both Mn. Body-ramus, Lt. scalloping, multilocular, Pano., Occ.
condyle, Symphyseal region expansion
6 1970 Morris et al.18 2 NA NA NA
7 1971 Huebner and 2 #23-26, #21-Lt. Mn. angle scalloping Pano., Peri.
Turlington19
8 1973 Stewart et al.20 2 #17-20, #30-32 scalloping Lat. view, Mn. PA
9 1974 Schofield13 3 #21-24, #19-20, #18-Lt. Mn. scalloping, multilocular, buccal Pano., Peri., Occ.
ramus erosion
10 1975 Ruprecht and Reid21 2 #20-22, #30-32 NA Peri.
11 1978 Heimdahl22 2 #30-22, #26-31 scalloping, expansion Pano., Occ.
12 1978 Pogrel6 2 #20- Lt. Mn. Ramus, #30-Rt. scalloping, expasion Pano., Mn. PA
ramus
13 1979 Markus5 2 #19-23, #25-29 multilocular, tooth displacement, Pano., Occ.
loss of lamina dura, root
resorption, expansion
14 1979 Raibley et al.23 3 #29-signoid notch, #24-25, scalloping, multilocular, Pano.
#19-20 expansion
15 1980 Kuroi24 NA All Mn. teeth NA Pano.
16 1981 Patrikiou et al.25 2 #19-22, #29-30 scalloping, expansion Pano.
17 1988 Forssell et al.26 2 #19-20, #29-31 scalloping NA
18 1991 Brannon and Houston27 2 #17, #32 (-) Pano.
19 1992 Saito et al.28 2 #17-18, #22-26 NA NA
20 1992 Saito et al.28 2 #18-19, #27-32 NA NA
21 1992 Saito et al.28 2 #10-11, #17-18 NA NA
22 1992 Prakash et al.29 3 #3-4, Mx. Ant. Region, #18-22 scalloping, expansion Pano.
23 1992 Fielding30 2 #19-22, #28-29 scalloping, expansion Pano., Peri.
24 1993 *Jones and Baughman7 3 Mn. Ant. Region, #20-ramus, expansion Pano., Occ.
#28-ramus
y
25 2002 Oda et al.31 2 #31-ramus, #27-Lt. ramus multilocular, tooth displacement, Pano.
loss of lamina dura, root
resorption, expansion
26 2003 Kraut and Robin32 2 #18-19, #29-31 scalloping, tooth displacement Pano.
27 2008 Mupparapu et al.33 3 #19-20, #17-18, #31-32 multilocular, loss of lamina dura Pano.
28 2010 Kuhmichel and 2 #30, #24-27 scalloping, expansion Pano., CT
Bouloux34
29 2012 de Oliveira et al.35 2 #21-25, #28-29 scalloping Pano.
30 2012 Martins-Filho et al.36 2 #18-21, #28-31 scalloping, expansion Pano.
31 2012 Mathew et al.37 2 #30-ramus, #19-20 scalloping, multilocular, Pano., CBCT
expansion
32 Our case 1 2 #22, #23-27 loss of lamina dura, expansion Pano., CBCT
33 Our case 2 2 #20-21, #28-30 scalloping, loss of lamina dura, Pano., CBCT
root resorption
34 Our case 3 2 #24-27, #29-30 scalloping, loss of lamina dura, Pano., CBCT
root resorption
ADA, American Dental Association; Pano., panoramic radiography; Occ., occlusal radiography; PA, periapical radiography; NA, not available; Mn.,
mandible; Mx., maxilla; Post., posterior; Ant., anterior; Lat., lateral; CT, computed tomography; CBCT, cone-beam computed tomography.
*Associated with osteogenesis imperfecta; asynchronous multiple SBCs (interval of 4 year 9 months).
y
Associated with idiopathic thrombocytopenic purpura; asynchronous multiple SBCs (interval of 1 year 10 months).

evident (Figure 8). The patient was referred to the Department
of Oral and Maxillofacial Surgery, Kyungpook National
University Dental Hospital for further assessment and treat-
ment. His medical history was unremarkable. He could not
recall any history of trauma to the mandible. Physical exam-
ination was likewise unremarkable, with no evidence of
swelling, asymmetry or lymphadenopathy. Intraoral exami-
nation disclosed no soft tissue abnormality or bony expansion
involving either side of the mandible. Electric pulp testing of
the adjacent teeth was within normal limits. CBCT imaging
using a 15 cm field of view at 120 kVp and 15 mA exhibited
the presence of bilateral moderately defined unilocular lesions
OOOO
Volume 117, Number 6
Fig. 1. Summary of 34 cases of multiple SBCs of the jaws. (A ¼ absent; F ¼ female; M ¼ male; NS ¼ not-stated; P ¼ present;
ts ¼ tissue.)
Fig. 2. Age distribution (N 30 patients).
(Figure 9). Slight thinning of the lingual cortex was noted on
the right side; however, any other effects on surrounding
structures were absent. The contents of the lesion were
homogenously dense, similar to or slightly less dense than
adjacent soft tissue. Both lesions were interpreted as most
likely representing SBCs on the basis of the radiographic
findings; although the low possibility of bilateral keratocystic
odontogenic tumors (KOTs) could not be ruled out. The
proposed treatment included exploration and incisional biopsy
of the both lesions. The lesions were approached surgically
under general anesthesia and noted to be empty without evi-
dence of a lining or fluid content. No tissue was available for
histopathologic examination. A diagnosis of bilateral SBCs
was established surgically and radiographically.
Case 3
An 18-year-old black male was referred to Graduate End-
odontics at the University of Michigan School of Dentistry for
endodontic therapy of the right mandibular second premolar.
His medical history was noncontributory and he could not
recall any history of trauma to the mandible. Physical exam-
ination was unremarkable, with no evidence of swelling,
asymmetry or lymphadenopathy. Intraoral examination dis-
closed no caries, periodontal disease or restorations in the
area. The vitality of the mandibular teeth, except for the right
CASE REPORT
An et al. e463
mandibular second premolar, was confirmed by electric pulp
testing. Periapical and occlusal radiographs revealed two
distinct unilocular radiolucencies involving the right
mandibular symphyseal region, extending from the left
mandibular central incisor to the right left canine, and the right
body of the mandible, extending from the distal aspect of the
area of the first premolar to the mesial aspect of the
second molar tooth (Figure 10). The right posterior mandib-
ular lesion was associated with root resorption of the second
premolar and mesial root of the first molar, loss of lamina
dura, and buccal-lingual bone expansion. The radiographic
impression was suggestive of SBC for the anterior lesion and
a suspected odontogenic cyst or benign odontogenic neoplasm
for the right posterior mandibular lesion. The proposed
treatment included incisional biopsy of both lesions with
endodontic therapy of the right mandibular second premolar
with Ca(OH)2 medication, apical Collaplug and Mineral
Trioxide Aggregate. Both lesions were approached surgically
under local anesthesia and were found to be empty, without
evidence of a distinct lining or fluid content (Figure 11).
Histopathologic examination of the right mandibular lesion
revealed thin fragments of fibrovascular connective tissue
without cyst lining, suggestive of simple bone cyst, and viable
bone consistent with the wall of simple bone cyst in the
anterior mandibular lesion (Figure 12). The postoperative
course was uneventful. A follow-up CBCT image (i-CAT
Imaging system, Next Generation, Imaging Sciences Inter-
national, Hatfield, US) at 3 months shows peripheral bony
healing of the right mandibular lesion (Figure 13).
DISCUSSION
The purpose of this study was to review previously
published cases of multiple SBCs of the jaws and
include three additional cases of our own in order to
analyze the characteristics of multiple SBCs. Suei et al.
reported that SBCs of the jaws have an equal preva-
lence in both genders,13 with most occurring in patients
ORAL AND MAXILLOFACIAL RADIOLOGY OOOO
e464 An et al. June 2014

Fig. 3. Distribution of multiple SBCs N ¼ 33 patients, Case no. 6 and maxillary anterior lesion of case no. 22 were excluded due to
unspecified information. (R ¼ right; L ¼ left; CC ¼ condyle-coronoid process; UR ¼ upper ramus; LR ¼ lower ramus;
MA ¼ mandibular angle; 8 ¼ third molar area; 7 ¼ second molar area; 6 ¼ first molar area; 5 ¼ second premolar area; 4 ¼ first
premolar area; 3 ¼ canine area; 2 ¼ lateral incisor area; 1 ¼ central incisor area.)

Fig. 5. Case 1. Panoramic radiograph from an11-year-old girl

Fig. 4. Number of lesions involving each region - N ¼ 33
patients, Case no. 6 and maxillary anterior lesion of case no.
22 were excluded due to unspecified information. (R ¼ right;
L ¼ left; CC ¼ condyle-coronoid process; UR ¼ upper
ramus; LR ¼ lower ramus; MA ¼ mandibular angle; 8 ¼ third
molar area; 7 ¼ second molar area; 6 ¼ first molar area;
5 ¼ second premolar area; 4 ¼ first premolar area; 3 ¼ canine
area; 2 ¼ lateral incisor area; 1 ¼ central incisor area.)
in the second decade of life.38 They are usually
asymptomatic and rarely expand the cortical plates.26,39
In the present study, we identified 34 cases of multiple
SBCs, comprising 10 males and 18 females (1.8:1,
female:male). The patient’s gender was not stated in 6
of the 34 patients. 52.9% of multiple SBCs cases were
identified in the second decade of life, with 23.5%
found in patients older than 30 years of age.
The most common radiographic finding in multifocal
SBCs was scalloping, consistent with what is tradi-
tionally described in single lesions.40 Bony expansion
reveals unilocular radiolucent lesions involving the left
mandibular canine and mandibular symphyseal regions.
and multilocular radiolucent patterns were observed in
15 (44.1%) and 7 (20.6%) patients respectively, which
is less common than what is reported in single SBCs.41
Generally, SBCs have a tendency to grow along the
long axis of the bone, causing minimal expansion;
however, expansion of the involved bone can occur and
is more common with larger lesions.40
The differential diagnosis of SBC includes KOT.42
KOTs affect the mandible 2:1 compared to the
maxilla,43 occurring primarily during the second and
third decades.44 KOTs tend to grow through the med-
ullary space with very little expansion and often have
scalloped borders similar to those of an SBC40,42;
however, the cortical boundary of the KOTs is more
defined, with adjacent teeth frequently resorbed or
displaced.40 Since nevoid basal cell carcinoma syn-
drome (NBCCS) shows multiple KOTs of jaws,45,46
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Volume 117, Number 6
Fig. 6. Case 1. (A) Panoramic reconstruction from CBCT shows moderately defined unilocular cystic lesions on the anterior
mandibular area. (B) Axial section of CBCT shows mild expansion of the buccal cortex and thinning of the lingual cortex.
Fig. 7. Case 1. Postoperative panoramic radiograph at
24 months reveals bony healing.
multiple SBCs need to be differentiated from NBCCS.
While most multiple SBCs involve in mandible bilat-
erally, the number of KOTs associated with NBCCS
can range from 1 to 28 (mean 5.1).45 Other diagnostic
criteria of NBCCS, such as basal cell carcinomas of the
skin, palmar or plantar pits, lamellar calcification of the
falx cerebri and rib anomalies are helpful in differen-
tiating this from multiple SBCs.46 In addition, there are
some radiographic similarities between multiple SBCs
and cherubism; however, cherubism develops in early
childhood, typically between 2 and 6 years of age.
Bilateral facial swelling, anterior displacement of teeth,
multilocular jaw lesions, and posterior epicenter in the
mandibular ramus are characteristic of cherubism.25
SBCs are diagnosed based on clinical, radiographic,
and surgical findings showing an empty cavity in the
bone without cyst lining.34,47 In this study, 64.7% of
multiple SBCs were asymptomatic, being discovered
on routine radiographic examination similar to what is
noted with single SBCs. Computed tomography
generally allows distinction between solid/fluid-filled
lesions and air-filled cavities. Suei et al.48 insisted that
the presence of gas in the cavity of SBC during surgery
has been erroneously interpreted. From their review of
52 computed tomography images, they were unable to
identify areas of lower density indicative of the pres-
ence of gas, whereas gas was reportedly confirmed at
surgery in 28 cases.48 Furthermore, a discrepancy
CASE REPORT
An et al. e465
Fig. 8. Case 2. Panoramic radiograph of a 17-year-old boy
shows a unilocular radiolucency in the left mandibular pre-
molar area and a scalloped unilocular radiolucency in the right
mandibular body area.
between magnetic resonance imaging and surgical
findings associated with the fluid in SBC cavities was
also reported. Rapid fluid absorption or varying
amounts of fluid within SBCs was proposed as a po-
tential explanation for this discrepancy.49 In this study,
7 of 11 patients reportedly presented with fluid at
aspiration but at the time of surgery fluid was noted
within the cavity in only one patient. Fifteen of the 23
patients revealed an air-filled cavity at surgery. Like-
wise, our three cases presented as empty cavities at
surgery, even though the cavities were interpreted as
being filled with cyst-like content in two cases based on
CBCT imaging. The reason for this discrepancy be-
tween radiographic and surgical findings is not fully
understood. Although precise determination of the
contents of the cavity may not be meaningful for the
treatment of SBC, correct interpretation is necessary for
an exact diagnosis and could be helpful in elucidating
the pathogenesis.41
To date, the pathogenesis of SBCs is far from being
established conclusively.41,50 The numerous different
proposed mechanisms give some insight into the lack of
understanding of this unusual entity. A widely accepted
etiology invokes traumatic injury, which results in
intramedullary hemorrhage, followed by failure of the
hematoma to organize and be replaced by hard tis-
sue10,51,52; however, no history of trauma could be
ORAL AND MAXILLOFACIAL RADIOLOGY OOOO
e466 An et al. June 2014

Fig. 9. Case 2. (A) Panoramic, (B) axial, and (C) coronal sections of CBCT reveal unilocular cystic lesion of the left mandibular
premolar and right mandibular body areas. Thinning of the right lingual mandibular cortex is observed on the axial plane section.

Fig. 10. Case 3. (A) and (B) periapical radiographs from an 18-year-old black male reveal a unilocular radiolucent lesion of the
mandibular symphyseal and the right mandibular body. (C) Occlusal view shows buccal cortical expansion and lingual cortical
thinning.

incorporation of synovial tissue.56 Others have sug-

Fig. 11. Case 3. An empty cavity was encountered in the
mandibular right lesion during surgical exploration.
elicited in many cases and the incidence of trauma in
patients with SBCs does not appear to be greater than
that in the general population.11 With the exception of
two patients, no history of trauma could be elicited from
our review of 34 cases. Furthermore, the presence of
bilateral lesions would lessen the possibility of trauma
as a causative factor, since traumatic events seldom take
place bilaterally. Other possible causes have been pro-
posed, including venous obstruction,53,54 faulty calcium
metabolism, low-grade infection,55 and intraosseous
gested that SBCs might be the end result of numerous
initiating factors.57,58 In this study, concomitant lesions
seen in cases of multiple SBCs included osteogenesis
imperfecta7 and idiopathic thrombocytopenic pur-
pura.31 In both cases, the traumatic bone cysts occurred
asynchronously. Jones and Baughman7 raised the pos-
sibility that asynchronous bilateral SBCs simply rep-
resented fortuitous findings and were not associated
with the more generalized skeletal abnormalities seen in
osteogenesis imperfecta. Oda et al.31 reported that not
only hemophilia but also other hemorrhagic conditions,
such as idiopathic thrombocytopenic purpura, can pre-
dispose to the development of hemorrhagic bone cysts.
There might be an underlying systemic component in
patients with multiple SBCs, such as predisposing to
vascular issues. This might also explain why women
are more predisposed, who tend to have more condi-
tions with vascular coagulopathies. This would be
supported by our observation that multiple SBCs
occurred asynchronously in two patients with underly-
ing conditions, osteogenesis imperfecta, and idiopathic
thrombocytopenic purpura, respectively. Bleeding di-
atheses are a well-known feature in patients with
osteogenesis imperfecta.59,60 There was no description
of underlying coagulopathies from our review, that’s
OOOO CASE REPORT
Volume 117, Number 6 An et al. e467

Fig. 12. Case 3. Histopathologic examination of the biopsied tissue. (A) Right posterior mandibular lesion. Photomicrograph
shows a thin strand of fibrous connective tissue with occasional chronic inflammatory cells, without epithelial lining. This made up
only a very small percentage of the contents of the lesion. Overall, the bony cavity was devoid of tissue (hematoxylin-eosin stain,
original magnification 30). (B) Anterior mandibular lesion. Histopathologic examination reveals dense viable cortical bone
devoid of underlying connective tissue or epithelial lining (hematoxylin-eosin, original magnification 40.)

good results.32,64 Spontaneous resolution of SBC has

Fig. 13. Case 3. Six months postsurgery. Panoramic recon-
struction of CBCT reveals bony healing at the periphery of the
right mandibular lesion.
because the clinicians might have no notation about it
and did not consider this as an underlying possibility;
therefore they did not order the correct tests or ask the
correct questions about medical history that might have
allowed them to identify these vascular issues.
In general, the mandibular body is by far the most
common location for SBCs, with the maxilla repre-
senting an uncommon site.11,34 One explanation for this
lower prevalence in the maxilla could be related to the
possibility that maxillary lesions are obscured by the
overall radiolucent appearance imparted by the maxil-
lary sinus.3,11,61 Another possible explanation could be
due to differences in the quality and amount of bone
marrow and vascularity in the maxilla compared to the
mandible.62 In this review, we noted that multiple SBCs
were most commonly located in the mandibular body.
Maxillary lesions were noted in only two patients, both
in association with mandibular lesions.
Curettage of the bone walls is the most widely
accepted approach to the management of SBCs of the
jaws, the goal being to promote the formation of a
blood clot and subsequent bone repair.41,63 Addition-
ally, application of Gelfoam,58 grafting of allogenic
bone with platelet-rich plasma and intralesional in-
jections of a mixture of blood, porous hydroxyapatite,
and bone fragments have been reported to produce
been described by several investigators, with periodic
radiographic follow-up recommended when the patient
declines a conventional surgical approach to treat-
ment.65,66 Some studies reported a low recurrence rate;
less than 2%, although this included many cases
without follow-up after treatment.19,24 According to
Suei at al.,4 the overall recurrence rate of SBCs of the
jaws was 26%, similar to that of extracranial SBCs
(29%).67 In the present study, 31 of 34 patients (91.2%)
were treated surgically and one patient exhibited reso-
lution of the lesions in the absence of any therapeutic
intervention at 4 years 5 months from the initial diag-
nosis. Recurrences after surgical intervention were
evident in only three cases; representing the three oldest
patients in the study group. Even though conservative
opening into the lesion and careful curettage of the
lining is generally sufficient for treatment, careful
follow-up is necessary for the evaluation of recurrence,
especially in older patients.
Extracranial multiple SBCs were reported in the
literature.68-70 In extracranial SBCs, four clinco-
anatomic varieties could be recognized: Classic meta-
physeal, nontubular, epiphyseal, and multiple.70
According to Abdel-Wanis and Tsuchiya,70 extracranial
multiple SBCs occur in the higher age group and show
very high male predominance. In their review of the
literature, 15 cysts (35.7%) were in nontubular bones
and six cysts (14.3%) were epiphyseal. Chigira et al.68
suggested that venous obstruction of the lesions play an
important role in the etiology of multiple SBCs based
on their treatment result. In the present study, even
though multiple SBCs of the jaws demonstrated some
differences from a single lesion, such as female pre-
dominance and higher incidence of bony expansion and
multilocular pattern, it is not sure that this is an another
entity.
ORAL AND MAXILLOFACIAL RADIOLOGY
e468 An et al.
In conclusion, knowledge of the clinical and radio-
graphic features in cases of multiple SBCs of the jaws is
helpful in accurately diagnosing this entity.
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Reprint requests:
Karp-Shik Choi, DDS, PhD, Chang-Hyeon An, DDS, PhD
Department of Oral and Maxillofacial Radiology
School of Dentistry
Kyungpook National University
2177 Dalgubeol-daero
Jung-gu
Daegu 700-412
Republic of Korea
ksichoi@knu.ac.kr; chan@knu.ac.kr