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www.eda-egypt.org • Codex : 70/1710
I.S.S.N 0070-9484
Oral Surgery
EGYPTIAN
DENTAL JOURNAL
Vol. 63, 3027:3034, October, 2017
* Assisstant Prof. in Oral & Maxillofacial Surgery Department
** Lecturer in Oral and Maxillofacial Surgery Department.
*** Lecturer in Oral Pathology Department.
**** Lecturer in Oral Medicine Department Faculty of Dentistry/ University of Tripoli
INTRODUCTION
TBC shows its non-neoplastic behavior and is
dened by World Health Organization (WHO) as
“an intraosseous cyst with fragile connective tissue
lining without any epithelium.(1) TBCs are also
referred to by various other names in the literature,
including extravasation cyst, hemorrhagic bone
cyst, progressive bone cavity, simple bone cyst,
solitary bone cyst, and unicameral bone cyst.(2)
The multitude of the names applied to this lesion
attests to the lack of understanding of the true
aetiology and pathogenesis.(3) TBCs were described
in 1929 (4) by Lucas and Blum as separate disease
entities.(5) Diagnostic criteria were established
TRAUMATIC BONE CYST OF THE MANDIBLE; DIAGNOSTIC
CHALLENGE AND MANAGEMENT. A CASE REPORT
Abeer Hussen Elsagali *, Nuri Mustafa Alarabi **,
Abdurahman Musbah Elmezwghi*** and Laila Omrou Hamad****
ABSTRACT
The traumatic bone (TBC) cyst is an uncommon benign empty or uid containing cavity
within bone that is not lined by epithelium. The etiopathogenesis of TBC is still unknown. TBC
is frequently encountered in young patients during the second and third decades of life. Sex
predilection is equal but some studies in literature suggest clear female predominance. Body of the
mandible between the canine and the third molar is the most common site (75%) in head and neck
region followed by mandibular symphysis. The cysts are usually asymptomatic. Associated teeth
are usually vital with no resorption or displacement. It expands the cortices and, seldom, intraoral or
extra oral swelling may be seen. Most of the TBCs are diagnosed incidentally in orthopantomogram
(OPG). On radiographic examination, a unilocular irregular but well dened lytic lesion is seen
characteristically extending between the roots of the teeth. TBC is representing approximately
1% of all jaw cysts. A nal diagnosis of a TBC is almost invariably made at the time of surgery,
where in identication of an empty air-lled cavity serves as a valuable diagnostic tool. Surgical
exploration was proved not only essential in making the right diagnosis but also curative from a
treatment plan perspective. Recurrence of TBC is assumed to be extremely rare. However, a distinct
proportion of recurrences may occur.
(3028) Abeer Hussen Elsagali, et al.E.D.J. Vol. 63, No. 4
in 1946, comprising of a single bony cavity
without epithelial lining, encompassed by bony
walls, lacking contents or containing liquid and/
or connective tissue.(6) Afterwards, Hansen added
another criterion i.e. upon surgery, the lesion is
essentially empty and occasionally the cavity may
contain some uid and/or small amount of tissues.(7)
WHO classied traumatic cyst as a non-neoplastic
osseous lesion due to absence of epithelial lining,
which demarcates it from other true cysts.(8) TBCs
are included in the group of bone-related lesions,
together with the aneurysmal bone cyst, ossifying
broma, brous dysplasia, osseous dysplasia,
central giant cell granuloma and cherubism.(9)
For explaining this lesion various theories
have been put forth by many authors,(10) none of
them explains all the clinical and pathological
characteristics of the lesion.(11) Regarding the
presence of a history of trauma in most reported
cases,(12) the traumatic–hemorrhagic theory seems to
be the most accepted.(13) The explanation underlying
the hypothesis of trauma(14) according to this
theory TBC arises from a focus of intramedullary
hemorrhage that causes a hematoma after trauma.
(15) This hematoma subsequently liquees and fails
to organize and be replaced with tissue.(16) The other
theories assert failure of growth and development
of osseous tissue, biochemical and mesenchymal
differentiation, degeneration of benign neoplasms
and low-grade infection.(17) According to Wakasa et
al. Florid cemento-osseous dysplasia is the initiative
factor for TBC to develop due to lymphatic drainage
obstruction.(18)
The TBC is an uncommon,(2) The lesion is
mostly diagnosed in patients below 30 years of
age with an approximate mean age of 20 years.(19)
Reduced prevalence of TBCs in older age group has
led to the speculation that this lesion may be self-
repairing.(20) Sex predilection is equal.(21) Although
some studies state they are more frequently in
men.(22) But some studies in literature suggest
clear female predominance (14:7).(23) The majority
of cases are seen in long bones (90%).(1) TBCs
are rare lesions, accounting for 1% of all jaw
cysts.(24) Most commonly, the TBC involves the
body or symphysis of the mandible,(25) with only
a few cases reported in the condyle.(26) Maxillary
lesions tend to be uncommon, although the reasons
for this are unclear.(27)
Most of times this lesion is asymptomatic and
discovered on routine radiographic examination
only.(28) However, dull pain is associated in 10–30%
of the patients,(7-15)
swelling, tooth sensitivity, and less
commonly with stula, root resorption, paresthesia,
displacement of inferior alveolar canal,(29) delayed
eruption of permanent teeth. Cortical plate
expansion is seen in some cases, which occurs
commonly on buccal side and pathological fracture
of the mandible.(30) The adjacent to the lesion teeth
are usually vital and there is no mobility.(31)
Radiographic features of TBCs are non-pathog-
nomonic.(6) However, on radiologic evaluation, the
lesion exhibits unilocular or multilocular radiolucen-
cy with well-dened or ill-dened margins. When
multiple teeth are affected the radiolucencies in-
volving the roots shows a dome like projections that
scallops between the roots(32) and often scallops be-
tween the roots of the teeth, almost always diagnos-
tic.(33) The scalloped outline, however, is often found
in edentulous areas also.(7) Furthermore, scalloped
margin is a sign of possible recurrence, although this
should not be confused with the interdental scallop-
ing associated with an intact lamina dura.(34)How-
ever, This radiographic pattern may vary, which
includes multilocular presentation, association with
impacted teeth, and multiple occurrences in the
same patient.(20) The histopathological evaluation
reveals a vascular connective tissue or a thick myxo-
bromatous proliferation intermixed with normal
bone,(35) with a scant yellowish, even blood colored
liquid,(36) devoid of epithelial lining in the edges of
lesion.(37) Numerous broblasts and giant cell like
osteoclasts are sometimes visible, with some newly
TRAUMATIC BONE CYST OF THE MANDIBLE; DIAGNOSTIC CHALLENGE (3029)
formed trabecular bone surrounded by numerous
osteoblasts. Numerous congested capillaries and
cholesterol crystals related to the osseous necrosis
also may be present. (38)
A nal diagnosis of a TBC is almost invariably
made at the time of surgery, when identication of
an empty air-lled cavity serves as a valuable diag-
nostic tool.(24) Diagnosis established with aspiration
of lesion and opening of bone window and accu-
mulation of bleeding and brin inside the cavity is
usually efcient for resolution of bone.(7) Because of
a lack of unique clinical and radiographic features,
it is important to establish the differential diagno-
sis between TBCs and other radiolucent lesions
of the jaws.(4) Differential diagnosis included cen-
tral giant cell granuloma, aneurysmal bone cysts,
haemangioma,(39) dentigerous cysts, keratocystic
odontogenic tumors, ameloblastomas, odontogenic
myxomas, calcifying epithelial odontogenic tumor,
adenomatoid odontogenic tumor, focal osteopo-
rotic bone marrow defect, intraosseous vascular
malformations, early stage of cementoma (peri-
apical cemental dysplasia), brous dysplasia and
cherubism.(21) Several treatment modalities have
been reported, including resection, curettage, bone
grafting, corticosteroid injection and more recent-
ly, injection of autologous medullary bone.(40) The
widely recommended treatment for TBCs is surgi-
cal exploration followed by curettage of the bony
walls.(23) It serves as both a diagnostic maneuver and
a denitive therapy by producing bleeding in the
cavity. Haemorrhage in the cavity forms a clot which
is eventually replaced by bone,(25) offering a good
prognosis.(23) It is believed that in some cases there
may be a spontaneous resolution,(41) that might al-
low more aggressive lesions keep growing, leading
to additional complications such as pathological
fracture, involvement of adjacent teeth and need for
wider surgical margins.(18)
Recurrence of TBC is assumed to be extremely
rare.(30) If there are multiple cysts recurrence rates
would be high(42) or those associated with orid
cemento-osseous dysplasia.(23)
Case Report
A 23 years old Libyan male was referred to the
Oral and Maxillofacial Surgery Department, Faculty
of Dentistry, University of Tripoli, for evaluation
and management of an asymptomatic unilocular
radiolucency in the posterior right mandible which
was discovered during routine OPG screening for
orthodontic treatment, with an insignicant medical
history.
On extra oral and intra oral clinical examination
no any pathological abnormality detected, the teeth
related to lesion were vital and immobile as well as
the facial symmetrical was observed.
On radiographic examination, OPG revealed
well dened unilocular radiolucent lesion measuring
approximately 3cm.x1.5cm. that extended from
the distal root of the right lower rst molar to the
distal aspect of right lower third molar (g.1), with
scalloped superior border between the roots (g.2).
Aspiration with 19 gauge disposable needle showed
negative result. On the basis of these ndings the
differential diagnosis was keratocystic odontogenic
tumor, ameloblastoma and traumatic bone cyst.
Fig. (1) Preoperative Orthopantomograph showing well dened
unilocular radiolucent lesion that extended from the
distal root of the right lower rst molar to the distal
aspect of right lower third molar.
(3030) Abeer Hussen Elsagali, et al.E.D.J. Vol. 63, No. 4
Surgical exploration procedure was done under
general anesthesia, intra-oral incision was done
extended from the right lower rst premolar to the
retromolar area, a mucoperiosteal ap was reected
and bone window was created bucally. Empty
bone cavity with small amount of serosanguineous
uid was found without any epithelial lining.
The entire bony wall was curetted carefully to
stimulate bleeding and to remove thin connective
tissue lining, irrigated well and closure of the ap
was done by using 4/0 absorbable suture. A small
amount of tissue fragments of viable bone and scant
soft tissues curetted was sent for histopathological
examination. The histopathologic result revealed
brous connective tissue, with interspersed bony
trabecular pattern without any epithelial lining,
few areas of hemorrhage and intense inammatory
cell inltrate (g.3). Correlation between clinical,
radiographic and histopathological result along
with the ndings of surgical exploration were
conrmed the nal diagnosis of the lesion was TBC.
Furthermore, the nal diagnosis was conrmed after
regression of the lesion as well as evidence of bone
regeneration with follow-up without recurrence
after one year (g.4 and 5).
Fig. (3) A photomicrograph showing brous connective tissue,
few areas of hemorrhage and intense inammatory cell
inltrate (H&E stain, x40).
Fig. (4) Six months follow-up orthopantomograph showing
new bone formation.
Fig. (5) One year follow-up orthopantomograph showing
regression of the lesion, progressive osseous
regeneration with no evidence of recurrence.
Fig. (2) Preoperative Orthopantomograph showing scalloped
superior border between the roots.
TRAUMATIC BONE CYST OF THE MANDIBLE; DIAGNOSTIC CHALLENGE (3031)
DISCUSSION
The TBC is an uncommon nonepithelial lined
cavity of the jaws,(43) characterized by an empty
bone cavity(11) or contain blood or straw-colored
uid.(1) In the presented case, empty bone cavity
with small amount of serosanguineous uid was
found without any epithelial lining, this nding
was found in most previous studies particularly
in the mandibular lesions. It usually occurs in the
metaphyseal region of long bones and it is unusual
in the maxillofacial region, with a prevalence
of 0.5–1.2% of all jaw cysts.(13) However, TBC
is uncommon and coincide with many previous
researches. TBCs are considered almost exclusively
mandibular lesions, with a preference for the
posterior areas (body and ramus),(44) 95% TBCs
occur in the body of mandible(8) or symphysis area
with only a few cases reported in the condyle.(24)
However, in our case the lesion was located in the
mandibular molars region in agreement with many
previous literatures. TBCs are generally detected
in patients in the second and third decades of life,
although in 15% it was found above 40 years of
age.(5) In the presented case the lesion was diagnosed
at the age of 23 years old which fall within
age range in which TBCs are most commonly
reported. The sex distribution is reported to be
quite even(31) or men are affected somewhat more
frequently.(45) our patient in this presented case
was male is inconsistent with Cortell-Ballester et
al 2009 who reported female predominant.(23) The
pathogenesis of traumatic bone cyst remains a matter of
controversy.(46) However, there are many etiological
theories postulated that has been suggested to elaborate
this lesion . Among them the mostly accepted theory
is the trauma hemorrhagic theory which explains its
clinical & histological features.(31) Nevertheless, the
traumatic etiology hypothesis is challenged by the
fact that more than 50% of cases have no traumatic
history.(7) Only 23.8% of the patients reported
antecedents of orofacial traumatism in the
retrospective study of Ballester et al.(23) Our
patient did not give any evident history of trauma.
Furthermore, the trauma-hemorrhage theory may
be an explanation because the mandible, which
has more cortical bone, repairs itself more slowly
compared to the maxilla. Clinically, the lesion is
asymptomatic in the majority of cases and(47) may be
incidentally diagnosed on routine dental treatment.(5)
our case was absolutely asymptomatic and coincide
with Paulo Ricardo et al study(2012) (4) and most
previous studies. Teeth vitality was notied in most
cases.(48) In our case report, the teeth involved were
vital and did not show root resorption, this nding is
in agreement with Lago CA et al 2006(49) and Neuschl
M. et al. 2014.(40) Radiographic examination usually
reveals a unilocular homogeneous radiolucency (50)
with a well-circumscribed margin.(51) our ndings
were similar and coincide with, Naveillie, Shaffer,
Lucas C, and Hansen. Characteristic for the TBC is
the “scalloping effect” when extending between the
roots of the teeth (52) as we found in our presented
case.
Most of the histopathologic ndings reveal -
brous connective tissue and normal bone. There is
no any evidence of an epithelial lining. The lesion
may exhibit areas of vascularity, brin, erythrocytes
and occasional giant cells adjacent to the bone sur-
face,(25) most of these features were evident in our
presented case and is in accordance with observa-
tion of MacDonald-Jankowski D(31), Kaugars(53) and
Hansen(7). Aspiration result in cases of TBC, a posi-
tive aspirate of straw colored uid is observed.(35)
The amount of uid diminishes with the age of the
lesion, and the lesion eventually becomes empty.(19)
In our case aspiration biopsy from the lesion showed
no cystic uid and air bubbles with some blood, this
ndings were coincided with L. K. Surej Kumar et
al. 2015.(5) and Kumar Pushpanshu et al. 2013.(24)
Diagnosis of Traumatic bone cyst prior to
surgical intervention is greatly difcult in most of
the instances.(5) Because of a lack of unique clinical
and radiographic features, it is important to establish
the differential diagnosis betweenTBCs and other
radiolucent lesions of the jaws.(24) Therefore, TBCs
can make differential diagnosis with aneurysmal
(3032) Abeer Hussen Elsagali, et al.E.D.J. Vol. 63, No. 4
bone cysts but (form very expansile soap-bubble
radiolucencies and usually in the mandibular
ramus and angle. Active and aggressive cysts tend
to recur), dentigerous cysts but (contain the crown
of an unerupted tooth which is usually displaced).
Keratocystic odontogenic tumors but (most lesions,
are unilocular, with as many as 40% noted adjacent
to the crown of an unerupted tooth and 50% of
mandibular lesions produce buccal expansion, with
a propensity for root divergence). Calcifying cystic
odontogenic tumor but (Because calcications are
usually small, lesions tend to occur as a diffuse
radiolucency with faint ecks calcied structures,
lesions have indistinct lines of demarcation with
the surrounding bone and usually occurs over
unerupted teeth it may be a radiolucent or mixed
unilocular lesion). Ameloblastomas but (It appears
radiographically as a multilocular radiolucency and
usually presents between ages 30 and 50 years old).
Odontogenic myxoma but (a radiolucent area with
a soap-bubble or honeycomb appearance and show
gross expansion of the mandible). Focal osteoporotic
bone marrow defect but (The radiographic
appearance is variable, ranging from a rounded,
barely noticeable mixed radiolucent-opaque lesion
to a diffuse, irregularly shaped radiolucent area with
faint wisps of a trabecular pattern). Intraosseous
vascular malformations but (usually diagnosed
with imaging studies such as Doppler angiography
or contrast time-lapse angiography). Central giant
cell lesions but (on occlusal radiographs, often
exhibit complete cortical bone loss, movement of
associated teeth and resorption of tooth roots is
commonly observed) and early stage of periapical
cemental dysplasia but (mostly black females are
affected and lesions are multiple and is considerably
more common in the anterior mandible).
The denitive diagnosis is mainly based on(49) the
analysis of clinical features, radiological features,
histological features and surgical exploration
ndings.(35) However, the clinical, radiographic,
histopathological ndings and surgical exploration
of the presented case that fulll the diagnostic
criteria, were enough to conrm the nal diagnosis
of TBC in the right mandible.
The widely recommended treatment for TBCs
is surgical exploration followed by curettage of
the bony walls.(54) Surgical exploration was proved
not only essential in making the right diagnosis,(55)
but also favors progressive bone regeneration after
formation of a stable blood clot, offering a good
prognosis.(23) In our case, surgical exploration
and curettage of bone walls was performed under
general anaesthesia.
Recurrence of TBC is assumed to be extremely
rare,(26) a recent study has reported the recurrence
rate of TBC in facial bone to be about 20%.(56) On
recurrence, a marked increase in the size with mul-
tilocular appearance.(57) If present, recurrence usu-
ally occur within three months after surgery.(20) Our
sixth month postoperative radiograph did not show
any recurrence and revealed excellent wound heal-
ing and in our presented case study simple curettage
of the walls of the bone cavity and surgical explo-
ration produced favorable results characterized by
regression of the lesion and progressive osseous
regeneration within 6 months duration with no evi-
dence of recurrence even one year after the surgical
treatment and our patient is still under observation
and follow up..
CONCLUSION
Diagnosis of TBC prior to surgical intervention
is a challenge in most of the instances, as the lesion
is asymptomatic and usually discovered on routine
radiographic examination for dental treatment tend
to be misdiagnosed with many tumors and cystic
lesions of the jaws. Surgical exploration followed
by careful curettage of the bony walls has been the
most widely recommended treatment for TBCs.
It serves as a diagnostic tool and as a denitive
treatment by inducing fresh bleeding which
undergo organization and followed by formation
of new bone. Clinical and radiological follow up is
obligatory to ensure complete osseous healing after
6-12 months and avoid possible recurrences.
TRAUMATIC BONE CYST OF THE MANDIBLE; DIAGNOSTIC CHALLENGE (3033)
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