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Journal of Cranio-Maxillo-Facial Surgery xxx (2012) 1e6

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Journal of Cranio-Maxillo-Facial Surgery

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Case report

Atypical presentations of simple bone cysts of the mandible: A case seriesand review of literature

José Ribamar Sabino-Bezerra, Alan Roger Santos-Silva, Jacks Jorge Jr., Adriele Ferreira Gouvêa,Márcio Ajudarte Lopes*

Oral Diagnosis Department, Semiology and Oral Pathology, Piracicaba Dental School, State University of Campinas, Piracicaba, Brazil

a r t i c l e i n f o

Article history:Paper received 15 May 2012Accepted 5 November 2012

Keywords:Intraosseous radiolucenciesSimple bone cystAtypicalMandible

* Corresponding author. Área Semiologia, FacuPiracicaba-UNICAMP, Avenida Limeira, 901, Bairro AreãSão Paulo, Brazil. Tel.: þ55 19 2106 5320; fax: þ55 19

E-mail address: malopes@fop.unicamp.br (M.A. Lo

1010-5182/$ e see front matter � 2012 European Asshttp://dx.doi.org/10.1016/j.jcms.2012.11.002

Please cite this article in press as: Sabino-Bezof literature, Journal of Cranio-Maxillo-Facia

a b s t r a c t

Simple bone cysts are well-defined intraosseous radiolucencies that often extend between the roots andappear clinically like empty cavities. This article aims to provide more information about this lesion withlimited prominence in academic literature, to illustrate atypical cases, and to provide a review of thecurrent literature. A series of six atypical cases of simple bone cysts is presented and their clinical,radiographic and microscopic characteristics, differential diagnosis, treatment and follow-up are dis-cussed. Correct diagnosis of this entity is of key importance, since it presents with clinical & radiographicsimilarities to other bone lesions, some exhibiting more aggressive behaviour.

� 2012 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rightsreserved.

1. Introduction

Simple bone cysts (SBCs) of the jawwere initially described as anentity in 1926 (Lucas and Blum, 1929), yet their diagnostic criteriawere only established in 1946 (Rushton, 1946). SBCs are classicallydescribed as well-defined radiolucent lesions, oftenwith scallopingof the margins between the roots of standing teeth. Clinically, theselesions demonstrate an empty cavity devoid of an epithelial lining,which are eventually filled with serous or sanguinous fluid(Rushton, 1946; Jundt, 2005). SBCs have been described usingdifferent terms in the literature, such as traumatic bone cysts,haemorrhagic bone cysts, extravasation cysts, solitary bone cysts,idiopathic bone cysts, idiopathic bone cavities and progressive bonecavities (Suei et al., 2007; Cortell-Ballester et al., 2009).

This intraosseous lesion is not unique to the jaw and is morecommonly found in long bones (90% of cases), mainly affecting themetaphyseal region of the proximal ends of the humeral (65%) andfemoral shafts (25%) (Lokiec and Wientroub, 1998). SBCs of the jawbone appear to be far less frequent (1%), and the body of themandible is usually affected (75%), mainly in the pre-molar andmolar regions (Copete et al., 1998). Maxillary lesionsmay also occur,but these are considered exceptionally uncommon (Mitchell andWard-Booth, 1984; Kuhmichel and Bouloux, 2010).

ldade de Odontologia deo, CEP 13414-903, Piracicaba,2106 5218.

pes).

ociation for Cranio-Maxillo-Facial

erra JR, et al., Atypical presenl Surgery (2012), http://dx.do

SBCs are most often diagnosed in the first 2 decades of life, witha slight femalepredominance, and are rarely seen after 25years of age(Tong et al., 2003). One important aspect is the fact that SBCs areusually asymptomatic, do not tend to cause bone swelling and theirdiagnosis often results from routine radiographic examination. Theradiographic pattern is described as a solitary, well-circumscribedradiolucent image with scalloped or irregular margins, which arewell-defined.Rarely, expansionof the cortical bone isnoticed (Forssellet al., 1988), and although highly suggestive, these image features arenot pathognomonic (Suomalainen et al., 2009). Even though thediagnostic criteria and radiographic features of SBC were previouslydefined, they do not always appear in a classic pattern. According toa few publications, the variations of SBCs can include a multilocularappearance, anassociationwith impacted teeth,multiple occurrencesin the same patient, asynchronous, and unusual sites (Forssell et al.,1988; Tong et al., 2003; de Souza Noronha et al., 2012).

Taking these considerations into account, SBCs could mimicother radiographic bone lesions, including those with moreaggressive behaviour, such as ameloblastoma, keratocystic odon-togenic tumour, myxoma, and central giant cell granuloma (Pitak-Arnnop et al., 2010; Shirani et al., 2011; Kansy et al., 2012).

Complementary examination, such as pulp vitality testing,computed tomography (CT) and biopsy, is therefore important forestablishing a proper diagnosis. Differential diagnoses includekeratocystic odontogenic tumours, central giant cell lesion, ame-loblastomas, myxomas and other odontogenic cysts and tumours.

Apparently, the association of the surgical finding of an emptycavity with the above clinical and radiographic characteristics is

Surgery. Published by Elsevier Ltd. All rights reserved.

tations of simple bone cysts of the mandible: A case series and reviewi.org/10.1016/j.jcms.2012.11.002

J.R. Sabino-Bezerra et al. / Journal of Cranio-Maxillo-Facial Surgery xxx (2012) 1e62

enough to establish the diagnosis of an SBC (Homem de Carvalhoet al., 2010). However, when material can be obtained during thesurgical procedure, histopathological analysis is mandatory andusually diagnostic.

As a result, the aim of this study is to report the details ofclinical, radiographical and surgical findings, as well as the outcomeand long term follow-up of six unusual SBCs affecting themandible.

2. Case reports

2.1. Case 1

A 16-year-old boy with temporomandibular joint disorder wasreferred to an orthodontist for treatment. On radiographic exami-nation, a multilocular radiolucent region involving the left poste-rior body and ascending ramus of the mandible was found. Thepatient was then referred to our clinic (Fig. 1A). Extension of thelesionwith marginal scalloping was noticed into the inter-radicularregions of the lower left molars. Even though the first, second andthird molars were involved, they were vital. The third molar wasimpacted in a mesioangular position, but the lesion had beenasymptomatic. Computer tomography revealed smooth boneexpansion and signs of perforation of the lingual cortex (Fig. 1B).With the provisional diagnosis of an aneurysmal bone cyst orameloblastoma, a fine needle aspiration was performed, but nomaterial was obtained. As a result, a surgical exploration wasexecuted and, after raising a mucoperiosteal flap, an empty cavitywas foundwith the inferior alveolar nerve lying in themiddle of theempty cavity. Microscopic examination of the overlying corticalbone showed small amounts of granulation tissue and no signs ofepithelial lining, with fibrousmaterial attached to the cortical bone.The final diagnosis was of SBC. Radiographic examination 3 monthsafter the biopsy showed reduction in size of the cavity, andevidence of total bone healing was detected at the 1 year follow-upappointment (Fig. 1C).

2.2. Case 2

A 19-year-old girl was referred to our clinic by her orthodontistwho had noticed the presence of a unilocular radiolucency on

Fig. 1. A. Panoramic radiograph showing a large radiolucent, multilocular lesion of the leexpansion and erosion of lingual cortex. C. Panoramic radiograph 1 year post-operative de

Fig. 2. A. Panoramic radiograph revealing a unilocular radiolucent, with scalloped, well-circuto the third molar. B. Panoramic radiograph 2 years post-operative showing complete bone

Please cite this article in press as: Sabino-Bezerra JR, et al., Atypical presenof literature, Journal of Cranio-Maxillo-Facial Surgery (2012), http://dx.do

radiographic examination. The radiolucent area had scalloped,well-circumscribed margins involving the left ascending ramus ofthe mandible and a relationship to the third molar (Fig. 2A). Noevidence of caries was seen, and the tooth was viable. No medicalhistory was considered relevant. Differential diagnoses included anameloblastoma, odontogenic keratocyst or giant cell lesion. Thethird molar was extracted and surgical exploration of the radiolu-cent area in the left mandibular ascending ramus detected anempty bone cavity that was carefully curetted around the cavitywalls. The bone tissue related to the lesion was taken for micro-scopic examination and showed normal cortical bone, fragments ofosteoid tissue and no signs of epithelial tissue. The final diagnosismade was that of a simple bone cyst. The patient then defaulted onfollow-up and was only located 2 years later. Radiographic exami-nation showed that the lesion had completely resolved (Fig. 2B).

2.3. Case 3

A 15-year-old girl was referred by her orthodontist to our clinicfor the investigation of a multilocular radiolucency extending fromthe right ascending ramus. The lesion had passed through the angleand body of the mandible and had reached the canine region, withmargins extending between the roots of associated teeth in a scal-loped and corticated pattern (Fig. 4A). There was a mild facialasymmetry on the right side (Fig. 3A). Intraorally, a slight boneswelling of the lingual and labial cortices was noticed (Fig. 3B). CTimages confirmed expansion of the buccal and lingual cortex of theright ramus and body of the mandible (Fig. 4B). The lesion wasasymptomatic and was found incidentally on radiographic exami-nation. There was no history suggestive of any underlying medicalproblem and all related teeth were viable. After the provisionaldiagnosis of an ameloblastoma, odontogenic keratocyst or centralgiant cell lesion, a needle aspirationwas performed and was shownto be negative for any material. Subsequently a surgical explorationwas performed of the involved region, revealing an empty bonycavity without any sign of cyst lining or capsule (Fig. 3C). Thematerial taken was submitted for histopathology. Analysis reportwas compatible with SBC (Fig. 3D). After 4 months of follow-up,a CT examination showed an increase in the degree of bonedensity of the lesion in the molar region compared with the

ft posterior mandibular body with scalloped margins. B. CT, revealing smooth bonemonstrating complete ossification of the defect.

mscribed margins involving the left ascending ramus of the mandible with relationshiphealing.

tations of simple bone cysts of themandible: A case series and reviewi.org/10.1016/j.jcms.2012.11.002

Fig. 4. A. Panoramic radiograph showing a large multilocular radiolucent of the right ascending ramus, and the angle and body of the mandible with scalloped and corticatedmargins. B. CT, showing expansion of the buccal and lingual cortex of the right mandibular ramus. C. Panoramic radiographic after 3 years of systematic follow-up, with noalterations detected.

Fig. 3. A. Extra-oral photograph showing facial asymmetry on the right side. B. Slight increase in volume in the buccal and lingual cortical. C. Intraoperative photo showing emptybone cavity. D. Microscopic features of simple bone cyst.

J.R. Sabino-Bezerra et al. / Journal of Cranio-Maxillo-Facial Surgery xxx (2012) 1e6 3

previous radiographs, as well as a reduction in size. Complete bonehealing was subsequently detected, and no alterations wereobserved after 3 years of systematic follow-up (Fig. 4C).

2.4. Case 4

A 13-year-old boy was undergoing the final phase of an ortho-dontic treatment when he was referred by his orthodontist to ourclinic for the investigation of a radiolucent, unilocular lesionextending from the left body of mandible to the canine region. Thelesion had irregular but well-defined margins and extendedbetween the roots of associated teeth in a scalloped and corticatedpattern (Fig. 5A). All of the left mandibular teeth were free of caries

Fig. 5. A. Panoramic radiograph showing unilocular radiolucent of the left posterior maradiograph demonstrating no alterations after 2 years of follow-up.

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and were still vital. With a clinical impression of ameloblastoma orodontogenic keratocyst, a diagnostic fine needle aspiration wasperformed with no fluid or liquid found. Afterwards, surgicalexplorationwas performed and an empty bony cavity encountered,so the diagnosis of SBC was concluded. Three months after thesurgical manipulation, good healing of the original defect wasobserved on radiographic examination, and complete bone healingwas detected after 2 years of follow-up (Fig. 5B).

2.5. Case 5

A 45-year-old womanwas referred by her general dentist to ourservice for investigation of a multilocular radiolucent region with

ndibular body with scalloped and corticated margins between roots. B. Panoramic

tations of simple bone cysts of the mandible: A case series and reviewi.org/10.1016/j.jcms.2012.11.002

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irregular margins in the right side of her mandible, which had beenfound incidentally on radiographic examination (Fig. 6A). CT,revealed a protuberant bone expansion with an irregular surface.Septum-like radiopaque lines are also seen (Fig. 6B). All mandibularteeth were viable. The clinical differential diagnoses included anameloblastoma, odontogenic keratocyst tumour or an odontogenicmyxoma. No content was found by fine needle aspiration, soa surgical exploration was executed. An empty bone cavity wasfound and bone tissue was taken for histopathological analysis.These results combined with the clinical features led to the diag-nosis of an SBC. After 6 months of follow-up, the original defect wasobserved to be healing well on radiographic examination (Fig. 6C).

2.6. Case 6

A 37-year-old woman was referred by her general dental prac-titioner for the investigation of a unilocular lesion with well-defined margins. This lesion was located in an edentulous regionbetween the second pre-molar and the second molar in her leftmandible, as her first molar was absent, and was incidentally foundon a routine radiographic examination (Fig. 7A). Tooth 36 wasextracted 16 years before she attended our clinic. Therewas no painor swelling associated with the lesion. Medical history revealedbreast cancer that had been treated by surgery and was awaitingadjuvant treatment. Differential diagnoses of the mandible lesionincluded a residual cyst, keratocyst odontogenic tumour andameloblastoma. A fine needle aspiration was performed prior to

Fig. 6. A. Panoramic radiograph revealing a multilocular radiolucent with scalloped and circCT, revealing a protuberant bone expansion with an irregular surface. Septum-like radiopaquthat good bone healing had occurred.

Fig. 7. A. Panoramic radiograph showing a unilocular radiolucent in an edentulous region

Table 1Summary of clinical features.

Case Age/gender Reference Rx

1 16/M Orthodontic evaluation Multilocular radiol2 19/F Orthodontic evaluation Unilocular radioluc3 15/F Orthodontic evaluation Multilocular radiol4 13/F Orthodontic evaluation Multilocular radiol5 45/F General dentistry Multilocular radiol6 37/F General dentistry Unilocular radioluc

Abbreviations: ABC, aneurismal bone cyst; AME, ameloblastoma; OKT, odontogenic kera

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surgical exploration, no fluid or liquid was found. Surgical explo-ration displayed an empty cavity, with insufficient material formeaningful histopathological analysis. The diagnosis of an SBC wasmade. Radiographic examination 6 months after the surgicalprocedure showed an increase in the degree of bone density andevidence of bone healing (Fig. 7B).

In this current case series, we believe the largest featuringatypical SBCs, several unusual clinical and radiographical aspectswere detected. Table 1 summarises the clinical features of the sixcases.

3. Discussion

There are many theories regarding the aetiology of SBCs, butnone these theories are able to explain the varied circumstancesunder which this entity is discovered. Despite the varioushypotheses in the literature, the aetiology of SBCs is still unclear.Among these theories, three predominate: an abnormality ofosseous growth, a degenerating bone process and an individualfactor triggering haemorrhagic trauma (Harnet et al., 2008). Themost striking observation to emerge from the data comparisonwas that trauma events are rarely involved (Tong et al., 2003; Sueiet al., 2007; Harnet et al., 2008), and the relevance of this asso-ciation is supported by our findings, as not all cases were associ-ated with trauma. We feel strongly therefore that thenomenclature of “traumatic bone cyst” should be no longer beused.

umscribed margins in the posterior mandibular body extending to the lower border. B.e lines are also seen. C. Radiograph taken 6 months after surgical exploration, indicating

. B. Panoramic radiograph revealing signs of bone healing 4 months after exploration.

Site Clinical hypothesis

ucent Posterior body ABC and AMEent Ascending ramus AME, OKT, and CGCLucent Posterior body and ascending ramus AME, OKT, and CGCLucent Posterior body AME and OKTucent Posterior body AME, OKT and CGCLent Posterior body (Edentulous area) RC

tocyst tumour; CGCL, central giant cell lesion; RC, residual cyst.

tations of simple bone cysts of themandible: A case series and reviewi.org/10.1016/j.jcms.2012.11.002

J.R. Sabino-Bezerra et al. / Journal of Cranio-Maxillo-Facial Surgery xxx (2012) 1e6 5

SBCs are most commonly observed during the second decade oflife (Saito et al., 1992; Tong et al., 2003) which is confirmed in themajority of our sample. However, the findings of the current seriesdo not support previous research asserting that atypical cases areusually associated with an older age group or with fibro-osseouslesions (MacDonald-Jankowski, 1995).

Comparing our data with the literature review by Tong et al.,2003, an interesting finding of two cases (case 5 and case 6) ofSBCs in an older age group that were not associated with cemento-osseous dysplasia (COD), including one in an edentulous area of themandible (case 6), which, to the best of our knowledge, has notbeen reported before. Such variation in the clinical presentation ofSBCs may be relevant to the better understanding of this under-explored lesion.

According to the literature (Copete et al., 1998), the mostcommon site for SBCs is the posterior body of the mandible, and,surprisingly, the ascending ramus. In English publications,however, SBCs involving the mandibular ramus are rare (Forssellet al., 1988; Shigematsu et al., 1994; Copete et al., 1998;Matsumura et al., 1998; Strabbing et al., 2011). Strabbing et al.(Strabbing et al., 2011) found 8 cases affecting the mandibularramus when reviewing 121 cases of SBC previously reported in theliterature. In the present series, cases 2 and 3 also involved themandibular ramus. These lesions showed radiographic character-istics that suggested differential diagnoses of ameloblastoma,odontogenic keratocyst tumour or a central giant cell lesion in viewof their location. Due to these radiographic characteristics, CTimaging is important for an accurate diagnosis (Mathew et al.,2012), as this may avoid unnecessarily aggressive surgicalapproaches, such as en bloc resections of themandible in the youngpatients affected by unusual SBCs.

Tong et al. (Tong et al., 2003) highlighted the fact that SBCsshould be included in the differential diagnosis of the multilocularradiolucent lesions associated with impacted teeth. Our findings incase 1 and 4 corroborate this suggestion, but our impression is thatit is important to emphasise the inclusion of simple bone cysts inthe differential diagnosis of radiolucent lesions of the posteriorbody of the mandible. However, these lesions are not associatedwith un-erupted teeth.

Interestingly, in four out of the six cases presented in our series,patients were referred for evaluation during pre-orthodontic treat-ment. There is scant information in the literature regarding the safetyof performing orthodontic treatment in patients with SBCs.Remarkably, inoneofourcases (case4), thepatientwas referredafterthe conclusion of orthodontic pre-treatment, as the patient was stillusing orthodontic devices when analysed by our clinical staff. Thiswas not apparently associated with a worse outcome of the bonelesion however, since the lesion showed complete bone healing aftersurgical exploration and diagnosis of the SBC.

Spontaneous resolution has been discussed by some authors(Damante et al., 2002; Cortell-Ballester et al., 2009), but in our casesthis was not considered a valid alternative as some lesions showmore aggressive behaviour (Cortell-Ballester et al., 2009; Homemde Carvalho et al., 2010). In our series, treatment consisted ofcareful curettage of the bone walls, with satisfactory results char-acterised by progressive bone healing and the absence of recur-rence in all cases. Most importantly, the systematic and carefulclinical and radiological follow-up is mandatory for both thera-peutic choices (Suei et al., 2007).

Suei et al. (Suei et al., 2007) found that most recurrences wereseen within 5 months of the surgical treatment, and this grouprecommended that follow-up should be continueduntil healingwasconfirmed radiographically within 3 years. In our sample, evidenceof newbone formation after surgerywas noticedwithin 3months ofthe surgery and complete healing was noticed radiographically

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between 6 months and 2 years. However, this varied depending onthe lesion size. Our opinion is that initial follow-ups should bemadewithin 5months of the surgery, as an interval of 1 year is sufficient toconfirm healing or recurrence, but less frequent examinationwouldbe more convenient for practitioners as well as patients. In caseswith COD follow-up examinations may be necessary even afterhealing because there is a high probability that new lesions coulddevelop in such cases (Melrose et al., 1976).

In only four of the six cases could material be obtained forhistological evaluation, which reinforces the fact that the absenceof soft tissue, such as epithelial tissue, is one of the most frequentfeatures of these lesions (Suomalainen et al., 2009; Homem deCarvalho et al., 2010).

4. Conclusions

Although the majority of SBCs present with similar clinical andradiographic features, some cases may be confused with other enti-ties. A number of the issues emerging from this finding relatespecifically to the importance of complementary examination, suchas radiographic exams, CT imaging, pulp vitality testing and biopsy,alongside surgical exploration, in order to establish an appropriatediagnosis of SBC. Based on the results of our case series, surgicalexploration with careful curettage of the bone walls can be consid-ered a satisfactory treatment choice for unusual SBCs, as this avoidsunnecessaryextensive surgery.Most importantly, in order to confirmthe success of treating these cases, systematic clinical and radio-graphic follow-up is mandatory. Our suggestion is that SBCs shouldbe included in the differential diagnosis of radiolucent lesions in themandible, either in the presence or absence of impacted teeth.

Conflict of interestNo conflicts of interest are present.

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tations of simple bone cysts of themandible: A case series and reviewi.org/10.1016/j.jcms.2012.11.002