CASE REPORT

Lateral periodontal cyst: report of case and reviewof the literature

Márcia de Andrade & Ana Paula Pantosi Silva &

Flávia Maria de Moraes Ramos-Perez & Yara Teresinha Corrêa Silva-Sousa &

Danyel Elias da Cruz Perez

Received: 10 August 2010 /Accepted: 17 November 2010# Springer-Verlag 2010

AbstractBackground As the lateral periodontal cyst (LPC) is anunusual odontogenic cyst, most papers are single casereports or series with a limited number of cases, withfew large series. The aim of this study is to report anadditional case of LPC, emphasizing the clinical, radio-graphic, and histopathological features, differential diagnosis,and review of 264 cases reported in the English-languageliterature.Case report A 51-year-old male patient presented with awell-delimited, radiolucent, mandibular lesion, locatedbetween the roots of the right lower lateral incisor andcanine and evidenced during routine radiographic examina-tion. A surgical excision was performed. Microscopically,there was a cystic cavity lined by simple squamousepithelium, compatible with LPC.Discussion LPC is an unusual odontogenic cyst andpresents a marked predilection for occurring in themandible between the roots of canines and premolars.Accurate clinical and imaging exams should be performedfor a correct approach and diagnosis.

Keywords Differential diagnosis . Lateral periodontalcyst . Review

Introduction

Lateral periodontal cyst (LPC) is an uncommon developmentodontogenic cyst, representing about 0.4% of all odontogeniccysts [1] and 0.7% of all cysts of the jaw bones [2]. Thislesion is defined as a radiolucent lesion that grows along thelateral surface of an erupted vital tooth, in which aninflammatory etiology has been excluded, based on clinicaland histological features [2, 3].

The LPCs originate from remnants of odontogenicepithelium [4, 5]. These lesions are more common in adultsduring the fifth to seventh decades of life and demonstrate amale predilection [5], despite the fact that some studieshave not reported a gender preponderance [2, 6–9]. MostLPCs are located in the mandibular–premolar area, followedby the anterior region of the maxilla [10–13]. Radiographicfeatures demonstrate a well-defined, circumscribed, round orovoid radiolucent lesion, usually with a sclerotic margin,preferentially localized between the apex and the cervicalmargin of the teeth [3].

Botryoid odontogenic cyst (BOC) is considered a variantof the LPC, presenting as a multicystic lesion. Due to thepolycystic aspect, radiographically, most of these lesionsare multilocular. In the same way, the gross aspect is similarto a cluster of grapes. This lesion may be extensive and hasa higher risk of recurrence than LPC [14, 15]. In bothlesions, LPC and BOC, the most adequate treatment is acomplete surgical enucleation [5].

As LPC is an uncommon lesion, most papers are singlecase reports or series with limited number of cases, withfew large series (Table 1). Thus, the aim of this study is to

M. de Andrade :A. P. P. Silva :Y. T. C. Silva-Sousa :D. E. da Cruz PerezSchool of Dentistry, University of Ribeirao Preto,Ribeirao Preto, Sao Paulo, Brazil

F. M. de Moraes Ramos-Perez :D. E. da Cruz PerezFederal University of Pernambuco,Recife, Pernambuco, Brazil

D. E. da Cruz Perez (*)Curso de Odontologia, Universidade Federal de Pernambuco,Av. Prof. Moraes Rego, 1235, Cidade Universitária,CEP: 50670-901, Recife, Pernambuco, Brazile-mail: perezdec2003@yahoo.com.br

Oral Maxillofac SurgDOI 10.1007/s10006-010-0257-2

report an additional case of LPC, emphasizing the clinical,radiographic, and histopathological features, differentialdiagnosis, and review of 264 cases reported in theEnglish-language literature.

Case report

A 51-year-old male patient was attended to in a privatedental clinic due to a radiolucent mandibular lesion,evidenced during routine radiographic examination. Thepatient denied pain or any other symptoms. On intraoralexam, there was a painless, well-circumscribed, slightswelling, sited in the gingival mucosa between the rightlower lateral incisor and canine, which presented a hardconsistency and was covered by normal mucosa.

Periapical radiography revealed a well-circumscribed,radiolucent, unilocular lesion, located in the mandible,laterally and between the roots of the right lower lateralincisor and canine, without corticated margins, measuringabout 1.0 cm in diameter. In addition, a slight divergence ofthe roots of the teeth was observed (Fig. 1). Thermal testrevealed pulpal vitality of the two teeth adjacent to thelesion. Additionally, periodontal exam excluded a lesion ofinflammatory origin. LPC and the keratocystic odontogenic

tumor were the most likely clinical and radiographicdiagnoses. Under local anesthesia, complete surgical excisionof the lesion was performed, without intercurrences.

Macroscopic analysis revealed a unicystic lesion. Histopath-ologically, the lesion consisted of a cystic cavity lined by simplenonkeratinizing squamous epithelium, although in someregions, the cavity was lined by a double layer of cells (Figs. 2and 3). Clear cells were also observed. Moreover, inflamma-tory cells were not observed in the connective tissue from thecystic wall. According to clinical, radiographic, and histo-pathological features, a diagnosis of LPC was established.After the treatment and adequate postoperative exams, thepatient was lost to follow-up.

Discussion

The LPC is an uncommon odontogenic cystic lesion of thejaws, which develops in the alveolar bone along the lateralsurface of a vital tooth [3, 5, 7, 16]. Most cases arediscovered on routine radiological examination, sinceusually, these lesions are initially asymptomatic, as it wasobserved in our case. However, the lesions can present agingival swelling during their development and growth [5,12, 13].

Authors Number ofcases

Mean age Gender(male/female)

Site(mandible/maxilla)

Cohen et al. [7] 37 54 18:19 29:8

Rasmusson et al. [27] 32 55 22:10 28:4

Carter et al. [8] 23 49.4 12:11 19:3a

Jones et al. [1] 28 48.2 16:12 NA

Shear and Speight [2] 24 Range 19–71 12:12 14:10

Table 1 Summary of theepidemiological features of largeLPC series

a The site was not available in onecase

Fig. 2 Cystic cavity lined by simple and double squamous epithelia.H&E, ×200, original magnification

Fig. 1 Well-circumscribed, radiolucent, unilocular lesion located inthe mandible between the roots of the right lower lateral incisor andcanine, with slight divergence of the roots

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In the present study, we reviewed the epidemiologicaland clinical features of 264 cases of LPC published in theEnglish-language literature [1–13, 16–29]. Moreover, thesedata were compared with that presented in the current case.Most cases occurred in patients between the fifth andseventh decades of life. The mean age of the available caseswas 50.8 years (ranging from 18 to 82 years) [1, 3, 5–9,11–13, 16–27], similar to the present case, considering that,in 71 cases, this aspect could not be evaluated in detail [2,4, 10, 28, 29]. The LPC presents a male predilection, with amale/female preponderance of 1.3:1, according to 221 caseswhere this information was available [1–3, 5–9, 11–13, 16–28]. However, some series did not found a genderpredilection [2, 6–9].

Regarding the site of the lesion, in 203 of 264 cases of LPCevaluated, this feature was recorded. Of these cases, 150(73.9%) were located in the mandible, whereas the maxillawas affected in 53 cases (26.1%) [1, 3, 5–9, 11–13, 16–18,23–29], as occurred in this case, which was sited in themandible. All series but one found a maxilla predilection forLPC [9].

Considering the cases located in the mandible, the mostaffected region is the premolar–canine–incisor area, mainlybetween the premolars [2, 3, 5–8, 11–13, 16–18, 23–26, 29].Of the evaluated mandibular cases, only five cases occurredin the molar region [3, 7, 9, 29]. As occurred in mostpreviously reported cases, the current case was locatedbetween the mandibular canine and lateral incisor. Now,based on the available maxillary cases, most of themoccurred in the anterior region [7, 18, 23, 29]. The maxillarypremolar and molar areas are rarely affected, with seven [7,12, 23] and two cases [7, 23] reported, respectively.

The radiographic appearance of the lesion is a well-circumscribed radiolucency, presenting a round or oval

shape and sclerotic margins, sited on the root lateral surfaceof vital teeth, mainly lower premolars [12, 24]. Neverthe-less, Senande et al. [9] reported a series of 11 cases, ofwhich eight occurred in the anterior region of the maxillaand presented an inverted pear-like image. Divergence ofthe roots of teeth is a common finding, but root resorptionhas not been documented [2, 30]. Although most of theLPCs did not reach more than 1.0 cm in diameter [4, 27],there are reports of lesions involving the entire lateralregion of the tooth root [5, 7, 9]. The occurrence of bilateralLPCs is very rare [21].

The differential diagnosis of LPC includes gingival cyst,lateral radicular cyst, keratocystic odontogenic tumor,pseudocysts, and radiolucent odontogenic tumors. Thegingival cyst is a rare soft tissue odontogenic cyst thatpresents similar epidemiological features to the LPC, with apeak frequency in the sixth decade of life, occurring mostcommonly in the mandibular premolar–canine region. Incontrast, the gingival cyst shows a slight female predilection[2, 31]. Particularly in LPCs that cause gingival swelling, agingival cyst should be excluded using adequate radiographicexamination and, eventually, with the transoperative finding[2, 13, 18]. The radicular cyst may develop along the lateralroot surface, being named lateral radicular cyst. This lesionoccurs due to pulp necrosis and an infected lateral accessoryroot canal or presents a periodontal origin [2]. The LPC mustbe distinguished from lateral radicular cysts in order to avoidunnecessary endodontic therapy. Sometimes, LPC is mis-diagnosed as a chronic lesion of endodontic origin [32].Thus, in all cases of radiolucencies located between roots,pulp vitality test and a careful periodontal inspection of theinvolved teeth should be performed. In the present case,detailed clinical and radiographic exams were carried out.

The keratocyst odontogenic tumor (KOT) occurs mostcommonly in the posterior region from the mandible,mainly in patients in their second and third decades of life,despite a peak frequency in the fifth decade that has beenalso reported [1, 2]. Although LPC is more frequent inolder patients, KOT comprises one of the main differentialdiagnoses of LPC, since 22.9% of the cases occur in theroot lateral surface [30]. Radiographically, the collateralKOT may present very similar features to LPC, and afterthe exclusion of an inflammatory origin, the lesion shouldbe surgically removed and sent for histopathologicalanalysis to confirm the definitive diagnosis [2, 30, 33].Other lesions have been reported in the root lateral surface,such as ameloblastoma and simple bone cyst, which mayshow similar features to LPC [33]. In the same way, thedefinitive diagnosis is established after histopathologicalanalysis or surgical exploration, as in simple bone cystcases.

Microscopically, LPC presents as a cystic cavity lined bya thin layer of epithelium and supported by a connective

Fig. 3 Cystic cavity lined by a double layer of epithelial cells. Noinflammatory cells were observed. H&E, ×400, original magnification

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tissue. Usually, no inflammatory cells are observed, althoughsome cases present scarce inflammation in the fibrous capsule.The epithelium is cuboidal to stratified squamous, non-keratinizing, as observed in the present case [4, 5, 7, 12, 13,26]. Frequently, there are epithelial thickenings or plaques, inaddition to the presence of many clear cells rich in glycogen,which are found either in plaques or in the superficial layersof the lining of epithelium [2, 3, 23]. In this case, althoughepithelial plaques were not found, clear cells were observedin superficial layers. Other excessively rare microscopicfindings have also been described, such as a case with anabundant amount of melanin in the epithelial cells [23] andanother that presented a squamous cell carcinoma arising inthe epithelial lining of the LPC [16].

BOC is considered a variant of LPC. Radiographically,most of them are polycystic (multilocular), but there areseveral unilocular cases reported. Different from the LPC,BOC usually are symptomatic, causing swelling, pain, andrarely, paresthesia [15]. Histopathological features of BOCpresent some differences when compared to those of LPC.The lesion is multicystic, showing septa of thin fibrousconnective tissue. The diagnosis of BOC, as in LPC, isbased on histopathological features [2].

Surgical enucleation is the most appropriate treatment forLPC, with preservation of the involved teeth, as it wasperformed in this case. Recurrence is rare [2]. In contrast,although the BOC cases are also treated by surgery, therecurrence rate of BOC may range between 18% and 30%[14, 15].

In conclusion, LPC is an unusual odontogenic cyst, mostfrequently found in men during the sixth decade of life, andpresents marked predilection for occurrence in the mandiblebetween the roots of canines and premolars. Accurateclinical and imaging exams should be performed for acorrect approach and diagnosis.

Acknowledgments Dr. Silva-Sousa and Dr. Perez are researchfellows of the National Council for Scientific and TechnologicalDevelopment (CNPq).

Conflict of interest The authors declare that they have no conflict ofinterest.

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