Published online: 2019-08-29 THIEME Case Report 99

Understanding Lateral Periodontal : A Case Report

Monica Roy Chandel1 Kundendu Arya Bishen2 Nikit Agrawal3 Himanshu Singh4

1Department of Oral and Maxillofacial , Index Institute of Address for correspondence Kundendu Arya Bishen, MDS, PhD, Dental Sciences, Malwanchal University, Indore, Madhya Pradesh, Department of Oral Pathology & Microbiology, Index Institute India of Dental Sciences, Malwanchal University, Indore 452016, 2Department of Oral Pathology and Microbiology, Index Institute of Madhya Pradesh, India (e-mail: [email protected]). Dental Sciences, Malwanchal University, Indore, Madhya Pradesh, India 3Department of Oral and Maxillofacial surgery, Index Institute of Dental Sciences, Malwanchal University, Indore, Madhya Pradesh, India 4Department of Oral Pathology and Microbiology, Index Institute of Dental Sciences, Malwanchal University, Indore, Madhya Pradesh, India

Dent J Adv Stud 2019;7:99–102

Abstract Lateral periodontal (LPCs) are developmental in origin and are typically seen in the canine- area in the mandible and less commonly in the maxilla. Reported rate of incidence of LPCs is less than 1%, and LPCs represent only 0.8% of entire central cysts of the maxillary bone. Despite its unique clinical and radiological presentation, it is finally diagnosed due to its unique histological characteristics. Here, we present one Keywords case with characteristic findings. The routine hematoxylin and eosin–stained sections ►►lateral periodontal revealed reduced enamel epithelium-like cystic lining that is made of thin, nonkera- cyst tinized stratified squamous epithelium along with some epithelial plaques. The clini- ►►pathogenesis of later- cal-radio-pathological correlation affirmed the diagnosis of LPC. The pathogenesis of

al periodontal cyst LPC has been discussed.

Introduction on lateral surface of root of teeth. The multicystic variant of LPC is known as botryoid (BOC) due to Lateral periodontal cyst (LPC) is a rare and strange form of resemblance of the macroscopic and microscopic features 1 developmental odontogenic cyst. Mandibular premolar with “bunch of grapes.” area appears to be the most common location for LPCs, but presence of this cyst on various other sites is also report- Case Presentation ed.2 LPCs appear to arise in intimate association with root surface (lateral) of erupted tooth.3 Among developmental A male patient, aged 50 years, visited to a dental practitioner odontogenic cysts, the incidence of LPC is quiet low. The with a chief complaint of swelling on cheek area and pain in mean age of occurrence is 52 years with predilection for area of the right lower back teeth. Clinical inspection revealed occurrence in age ranges from fifth to seventh decades and a well-circumscribed and a well-defined ovoid swelling of no predilection for race or sex.4 Histopathologically, LPCs size 2 cm × 1 cm on the lower right canine to premolar region are classified under developmental cysts with lumen lin- (►Fig. 1). The swelling was nontender in nature, and there ing showing thin, nonkeratinized epithelium that is mostly was absence of any pulsations. Clinically the teeth were non- one to five cell layers thick—resembling reduced enamel carious and vital. epithelium. The epithelial lining illustrates focal thicken- Radiographic examination affirmed a well-defined ings or plaques where clear glycogen containing epithelial radiolucency (pear-shaped) in the upper and middle third cells are seen. The underlying to the epi- of 43 and 44 with a sclerotic border. Also, interdental bone thelium exhibits zone of hyalinization.5 Radiographically, loss was clearly evident in between them (►Fig. 2). Based a well-delineated ovoid or round radiolucent area with a on aforementioned findings, a diagnosis of LPC was given sclerotic margin and maximum diameter of 1 cm is seen clinicoradiologically.

received DOI https://doi.org/ ©2019 Bhojia Dental College and April 30, 2019 10.1055/s-0039-1693093 Hospital affiliated to Himachal accepted ISSN 2321-1482. Pradesh University May 20, 2019 published online August 29, 2019 100 Understanding LPC Chandel et al.

Histopathology examination of the tissue capsule was done. The hematoxylin and eosin–stained sections revealed a single cavity lesion lined by two to three layered squamous epithelium. In the epithelial lining, some clefts were seen and few areas formed of thick cluster of cells in midst of which clear cells were also observed (►Fig. 3). The connective tissue capsule was fibrous, and was present in few places. The histopathological findings confirmed the clinical diagnosis of LPC.

Discussion

LPC is a bizarre but well-recognized form of developmental cyst of odontogenic origin.3 The LPCs amount for less than 2% of all jaw cysts, which are epithelium lined. LPCs most com- monly appear in patients in their fifth or seventh decade of life; barely it occurs in patients younger than 30 years. The most common location seen is premolar region of the man- dible, followed by the maxillary anterior region.1 Fig. 1 Photograph showing swelling on right canine-premolar area. Clinically, LPCs appear to be symptomless and are acci- dentally detected during routine radiological investigation. Occasionally, if cyst is located on the labial surface of the root, a slight swollen mass may be obvious, although the overlying mucosa usually appears to be normal. Unless oth- erwise infected, the associated tooth is always vital. If the cyst becomes infected, it may resemble a lateral periodontal abscess. Radiological examination reveals that the cyst develops

as a well-circumscribed radiolucent area placed laterally to roots of a vital tooth. Most of them are less than 1 cm in their greatest diameter.1 In most cases, the border is definitive and is even surrounded sometimes by a thin layer of sclerotic bone.3 The origin of LPC is quite debated. Demonstration of ori- gin of LPC from any specific source is lacking, and therefore any hypothesis about its origin must be established on base of presumptive evidence. Histopathological studies have revealed that LPCs are usually deficient of inflammatory

Fig. 2 Radiograph showing cystic lesion between the roots of pre- molars in middle third area.

The treatment plan comprises total surgical enucleation of cyst. Local anesthesia was administered, and after achiev- ing the desired effect of local anesthesia, mucoperiosteal flap with full thickness was elevated. The cystic capsule was

detached from the neighboring bone, and a complete enucle- Fig. 3 Photomicrographs of histologic sections of cyst showing ation of the lesion was done using a surgical curette. The cyst ­cavity lined by two to three layered squamous epithelium with some capsule was sent for microscopic evaluation. clefts and few areas with thick cluster of cells.

Dental Journal of Advance Studies Vol. 07 No. 2/2019 Understanding LPC Chandel et al. 101 component. Though it may be present at a distance from the proximity. According to them, if such a case is left untreat- lining, it is suitable to regard them as actuality of develop- ed, these multiple LPCs can fuse to form a larger­ multicystic mental origin.6 LPC seems to be derived from either reduced lesion that is referred as botryoid variety.9 Altini and Shear enamel epithelium or cell rests of Malassez or remnants of (1992) stated that with continuous growth, LPC can take on dental lamina. botryoid (grape-like) appearance. They proposed a hypothe- According to the histopathological description, most of sis about “a unicystic LPC progressing to a multicystic-encap- the part of the cyst lumen is lined by a definite nonkerati- sulated lesion, and then how the progressive enlargement nized epithelium that may be reduced enamel epithelium, of the multiple microcysts lead to development of botryoid and hence there may be a possibility of origin of cyst from it.6 cyst—an irregular thin-walled multicystic structure.”5 On the other hand, Wysocki et al (1980) have proposed that They categorized LPCs into three types, on basis of the LPCs may have originated from clear cells of dental lami- morphology: na. They proposed that the LPC is derived from postfunction- al cells of dental lamina. They emphasized that the clear cells •• Unicystic consisting of glycogen may be demonstrated in the remnants •• Multicystic of dental lamina and that identical cells also appear in parts •• Botryoid of LPC lining and also in the epithelial plaques of LPC lining.7 Contradicting this, Van der wall stated that BOC cannot be Another proposed possibility of origin of LPC is that it arises contemplated as LPC variant because it extends beyond the from the remnant cells of Hertwig epithelial root sheath (cell root (lateral area), though he did not oppose that the possi- rests of Malassez). These cell rests of Malassez are present in bility of origin of cells for both cysts are same.10 from where they may get arranged in LPC. The If the lesion is found to be unilocular on radiological exam- support for this theory of origin is insufficient.6 ination, LPC is managed by surgical enucleation. Though sac- Histologically, the LPCs are described as cysts with non- rificing of the tooth involved is avoided, but it is not always keratinized thin (one to five cell layers thick) epithelial lin- possible. Recurrence is not a common feature of LPC, but it is ing, which closely coincide with the appearance of reduced noted in case of botryoid variant.3 enamel epithelium. Cuboidal or columnar epithelium may also be seen constituting the lining of cyst.3 Focal aggregates of glycogen-rich clear cells may be found amidst the epithelial Conclusion cells of cyst lining. Characteristically, few cysts present with LPCs are rare and an uncommon odontogenic cyst. Although focal nodular thickenings of epithelium lining, which mainly periodically reported, chances of their recurrence are rare. 1 comprises clear cells. Also, sometimes clear cells epithelial During enucleation of cyst, appropriate care must be taken rests are seen within the fibrous walls. A striking feature seen not to destruct the root of neighboring teeth. For the definite in number of LPC cases is the presence of localized epithelial diagnosis of LPC, the histopathological examination of entire line thickening or plaques. Sometimes the epithelial plaques specimen is important. may be small; other times they may be larger and may pro- trude into the encompassing cyst wall producing mural Note ­bulges. These plaque cells are fusiform having their long axes Prior to this publication, this study was not published parallel to the basement membrane. Generally, they are clear, ­anywhere else. 6 large as well as with small and pyknotic nuclei. Funding Based on the histological study at the light microscopy None. level, the usual sequence of development appears to be that there is proliferation of flat basal cells that produces a modest­ Conflict of Interest localized epithelial thickening. Though the actual reason None declared. behind these localized epithelial thickenings is not acknowl- edged, it seems to be the result of an instinctive process that References influences the lesions of the odontogenic epithelium and in 1 Neville D, Allen B, Textbook of Oral and Maxillofacial ­Pathology. 6 reduced enamel epithelium. 3rd ed. Saunders: Elsevier; 2009:692 According to Weathers and Waldron, BOC is a polycystic 2 Krier PW. Lateral periodontal cyst. Oral Surg Oral Med Oral type of the LPC, named so because the sample resembles an Pathol 1980;49(5):475 array of grapes. It is noninflammatory, developmental odon- 3 Shafer R, Rajendran SB. Textbook of Oral Pathology. 7th ed. India: Elsevier; 2012:269. togenic cyst having predilection for occurrence in the lower 4 de Carvalho LF, Lima CF, Cabral LA, Brandão AA, Almeida JD canine to the premolar region. Swelling with pain and par- Lateral periodontal cyst: a case report and literature review. esthesia is a frequent symptom shown. BOCs are larger cysts J Oral Maxillofac Res 2010;1(4):e5 than LPCs, their size ranges from 4 to 4.5 mm, and they can 5 Altini M, Shear M. The lateral periodontal cyst: an update. be unilocular to multilocular.8 They exhibit thin connective J Oral Pathol Med 1992;21(6):245–250 tissue septae, which is lined by thin nonkeratinized stratified 6 Shear M, Speight P, Cysts of the Oral and Maxillofacial Regions. 4th ed. Denmark: Munksgaard Blackwell;2007:84 squamous epithelium.8 Redman et al (1990) postulated that 7 Wysocki GP, Brannon RB, Gardner DG, Sapp P. Histogenesis of the BOCs may show multicentric origin. They supported­ the the lateral periodontal cyst and the of the adult. theory that in some patients, multiple LPCs develop in close Oral Surg Oral Med Oral Pathol 1980;50(4):327–334

Dental Journal of Advance Studies Vol. 07 No. 2/2019 102 Understanding LPC Chandel et al.

8 Weathers DR, Waldron CA. Unusual multilocular cysts of the 10 Arora P, Bishen KA, Gupta N, Jamdade A, Kumar GR. jaws (lateral periodontal cyst). Oral Surg Oral Med Oral Pathol ­Bortryoidodontogenic cyst developing from lateral periodon- 1973;36:245–41 tal cyst: a rare case and review on pathogenesis. Contemp Clin 9 Redman RS, Whitestone BW, Winne CE, Hudec MW, Patterson Dent 2012;3(3):326–329 RH. Botryoid odontogenic cyst. Report of a case with histologic­ evidence of multicentric origin. Int J Oral Maxillofac Surg 1990;19(3):144–146

Dental Journal of Advance Studies Vol. 07 No. 2/2019