https://doi.org/10.5272/jimab.2018242.2065 Journal of IMAB Journal of IMAB - Annual Proceeding (Scientific Papers). 2018 Apr-Jun;24(2) ISSN: 1312-773X https://www.journal-imab-bg.org Case reports

GINGIVAL OF ADULTS- TWO CASE REPORTS AND LITERATURE REVIEW

Elitsa Deliverska1, Aleksandar Stamatoski2 1) Department of Oral and Maxillofacial , Faculty of Dental Medicine, Medical University – Sofia, Bulgaria. 2) Department of maxillofacial surgery, Faculty of Dental Medicine, Ss. Cyril and Methodius University- Skopje, Macedonia.

ABSTRACT usually found in the incisor, canine, and areas. Background: of adult is an [1, 3, 4] uncommon, small, non inflammatory, extra-osseous, Clinically, the gingival may certainly occur developmental cyst of gingiva arising from the rests of without involvement and may appear as painless, . small sessile soft tissue swellings, usually involving the Purpose: The aim of our paper is to present two rare interdental area of the attached gingiva. clinical cases of gingival cyst of adult. These lesions measure about 0.5 to 1 cm in diameter. Material and methods: In the present cases, the They are often bluish or blue-gray due to thinning of the combined anatomic characteristics of the soft tissue overlying mucosa. In some instances, the cyst may cause presentation and the osseous defect suggest that the lesion slight erosion of the surface of the bone, which is usually is a gingival cyst of adult. Two cases of gingival cyst were not detected on a radiograph but is apparent during surgical diagnosed and treated with exicisional biopsy followed by exploration. histopathological confirmation and an emphasis on the If more bone is missing, one could argue that the clinical aspects of this lesion. lesion may be a that has eroded the Results: The biopsy revealed histologic features cortical bone rather than a gingival cyst that originated in consistent with a gingival cyst. Clinical and radiographic the mucosa. examinations 1 year post-surgery indicated uneventful soft The gingival cyst of adults has no radiographic tissue healing and bone fill of the initial defect. evidence of bone resorption, and only a reduced KVP Conclusion: Gingival cyst most commonly occurs on potential exposed periapical radiograph will show its soft the labial side of the in the canine and premolar tissue density. Where the radiolucency is dark and sharply areas. It is discrete, well-cirumscribed, painless swelling of demarcated, then communication with the is he attached gingiva or gingival papilla. This is a soft tissue indicated, and the lesion is more likely to be a lateral lesion, and if enlarged to sufficient size, it causes periodontal cyst that has eroded outwards. [3, 4, 5, 6] superficial erosion of the cortical plate of bone. Lesions appear to be asymptomatic and do not produce discomfort. Histopathologic features It is a fairly rare cyst or at least it is rarely sent for biopsy. The diagnosis of gingival cyst of the adult should be restricted to lesions with the same histopathologic Key words: , excisional biopsy, features as those of the lateral periodontal cyst. gingival cyst The lining is usually thin, resembling reduced enamel epithelium, and may have plaque-like thickenings. INTRODUCTION I Small nests of these glycogen-rich clear cells may also be The gingival cyst of the adult is an uncommon present. Sometimes the lining may be a stratified squamous lesion. It is an odontogenic developmental cyst of the epithelium without rete ridges, and occasionally it is gingival soft tissue derived from the rests of dental lamina keratinized. [2, 3, 6] that occurs primarily in middle aged adults. The condition is more frequent in patients over 40 years of age. [1, 2] Differential diagnosis include lateral periodontal cyst, , mucocele, fibroma, peripheral Clinical and radiographic features ossifying fibroma, neurofibroma etc. Gingival cysts of adults are most common in the canine and premolar area of the mandible (60% to 75% of The gingival cyst of the adult responds well to simple cases) and usually occurs during the fifth and sixth decades surgical excision. The prognosis is excellent. Recurrence of life. They are almost invariably located on the facial is not seen. gingiva or alveolar mucosa. Maxillary gingival cysts are In this paper, we describe two cases of gingival cyst

J of IMAB. 2018 Apr-Jun;24(2) https://www.journal-imab-bg.org 2065 of the adult, located in the alveolar mucosa on the facial radiographical finding suggesting of osseous involvement; aspect of the mandibular lateral incisor-canine/ canine -first peripheral fibroma was ruled since the lesion was soft and premolar region. [7, 8, 9, 10] cystic in consistency and odontogenic was ruled out since the lesion was not associated with pain or CASE REPORTS localized expansion of bone, and again radiographically Case 1 there was no osseous involvement. The treatment plan 45-year-old female patient presented at the included scaling, root planning, re-evaluation, and Department of Oral and Maxillofacial surgery, Medical excisional biopsy of the lesion. Anesthesia was obtained University - Sofia, with a complaint of a swelling in the with local infiltration. Excision of the lesion was performed gum on the left side of the lower jaw dating from six to with scalpel. The surgical specimen measuring 5 × 5 mm seven months prior to presentation. The gingiva in this area was placed in 10% neutral buffered formalin. After removal was apparently normal before the appearance of the lesion of lesion, the area was irrigated with sterile saline and according to patient. She could not recall any recent Iodine povidon 10 % solution. We achieved primary trauma, pain, or discharge from the swelling or an increase closure secured with a 3-0 black silk suture. Gentle pressure in the size of the lesion. Medical history was non- was applied with wet gauze to achieve hemostasis, and the significant, and the patient was not on any medication at wound was covered with a Parodium gel. The patient was that time. given postoperative instructions and was dismissed with During intraoral examination, a smooth round prescription for an analgesic (tab Ibuprofen-400 mg every nodule approximately 5 mm in diameter was observed. It eight hours), antibiotic (Amoxicillin-1,0 g - 2x1 for five was located on the attached gingiva in relation to the lower days) and antimicrobial rinse (Tantum verde spray- twice a left canine and lateral incisor extending inferiorly up to day for one week). Postoperative period was without any the mucogingival junction. complications.

Fig. 1. Clinical presentation of the lesion in frontal Case 2 region of lower jaw gingiva. A 64 years old male patient referred to our department for multiple tooth extractions. Prior to this visit, the patient had not received dental care for more than 10 years. During the intraoral examination, a lesion with clinical characteristic of gingival cyst was found on the vestibular mandibular gingiva between 33 and 34 teeth (fig.2).

Fig. 2 Clinical presentation of the lesion.

The nodule was non - tender, soft, and cystic in consistency, fluctuant and non-compressible (fig.1). Probing of the adjacent teeth yielded pocket depths of 2-3 mm with all sites exhibiting bleeding on probing, without any communication between the sulci of the adjacent teeth and the lesion. Pulp testing of the canine and lateral incisor indicated that both were vital. Radiographically, there was no finding suggestive of osseous involvement. Based on the clinical and radiographic findings, the provisional The lesion was nodular with a sessile base, diagnosis of cystic lesion of the gingiva was made. The nonulcerated, or painful; it was normal in colour and differential diagnosis included a lateral periodontal cyst, measured 5.0 mm in diameter. An excisional biopsy under peripheral fibroma and . Lateral local anesthesia was performed, and surgical exploration periodontal cyst was ruled out because there was no revealed osseous surface erosion. (fig. 3, 4)

2066 https://www.journal-imab-bg.org J of IMAB. 2018 Apr-Jun;24(2) Fig. 3. Intraoral radiological examination of the DISCUSSION same patient. The presented cases documented examples of gingival cyst of adult treated with excisional biopsy. The outward appearance of a gingival cyst of adult is typically an oval-to-round, firm, elevated swelling located on the attached gingiva. Commonly they are <5 mm in diameter, but lesions >5 cm can also be seen.[3] The lesions often demonstrate little or no change in colour in the overlying mucosa, but some appear bluish due to the cystic fluid. Eighty percent are found in the mandible, with the most common site being the canine-premolar area. The gingival cyst of adult is believed to be of odontogenic derivation, specifically from the remnants of ectodermic tissue.[11] The gingival cyst of adult is not related to any inflammatory lesion, and the associated teeth when tested are vital. The radiograph does not accurately depict the topography of this soft tissue-originating cyst, as the cyst may involve the alveolar bone surface and can often compromise the buccal Fig. 4. Osseous surface resorption under the lesion or lingual plate with a shallow 2 2 saucer-like2 2 defect. [12] Root exposure associated with this lesion is an extremely rare finding. Gingival cyst of adult is routinely treated with excisional biopsy and has a rare chance of recurrence. The differential diagnosis of gingival cyst of adult includes several lesions presenting as gingival swellings such as a lateral periodontal cyst and peripheral fibroma. The lesion that may be more difficult to differentiate is the lateral periodontal cyst, especially in the presence of both radiographic and gingival involvement. [3, 14, 15] This is because both lateral periodontal cyst and gingival cyst of adult are non- inflammatory cystic lesions, typically present in the mandibular canine-premolar area, and share similar histopathological characteristics.[5, 16] In the present cases, the combined anatomic characteristics of the soft tissue presentation and the osseous destruction in second case suggest that the lesion was gingival cyst of adult. Histopathological analysis confirmed the clinical There is a great deal of confusion about the hypothesis of gingival cyst. Postoperative period was relationship between the gingival cyst of adults and the uneventful with primary healing of the defect (Fig. 5). lateral periodontal cyst, much of which appears to have arisen because both types of cyst have a predilection for Fig. 5. Postoperative status 7 days after surgery. occurrence in the canine and premolar area of the mandible and, less frequently, in the maxilla. The pathogenesis of these cysts, particularly with regard to the cells of origin, is also far from clear. On the basis of the clinical and morphological similarities between the two cysts, it is believed that they have a common histogenesis and probably the same epithelial origin and that they represent the intra-osseous and extra-osseous manifestations of the same lesion. [14, 15, 17, 18, 19] It is considered to represent the soft tissue (the extra- osseous) counterpart of the intra-osseous lateral periodontal cyst. Gingival cysts of the adult are thought to arise from dental lamina rests or from entrapment of surface epithelium. [5,13]

J of IMAB. 2018 Apr-Jun;24(2) https://www.journal-imab-bg.org 2067 CONCLUSION The gingival cyst of adult is a unique pathologic biopsy is definitive. Exposure of the tooth root is an lesion of the oral cavity, typically localized in the extremely rare feature of the gingival cyst of adult; in such mandibular canine and premolar region, appearing in adults cases, as in the present report, a combined regenerative in their fourth to fifth decades of life. Associated osseous treatment approach may be used to achieve resolution of involvement occurs <50% of cases and is often the lesion-associated osseous defect and the excisional undetectable radiographically. Treatment by excisional biopsy-associated soft tissue defect.

REFERENCES: 1. Greenberg M, Glick M, Burkett’s 2010. 15. Hegde U, Reddy R. Gingival Diagnosis and treat- 8. Fragiskos DF. Oral surgery. cyst of adult—a case report with unu- ment; tenth edition; 2003 BC Decker Springer. 2007 sual findings. Indian J Dent Res. 2004 Inc. 9. Peterson’s principles of oral and Apr-Jun;15(2):78-80. [PubMed] 2. Cawson RA, Odell EW, Porter S, maxillofacial surgery. Second Edition. 16. Giunta JL. Gingival cysts in Cawson ‘s essentials of oral Miloro M. Editor. BC Decker Inc. the adult. J Periodontol. 2002 and oral medicine, Seventh edition, 2004. Jul;73(7): 827-31. [PubMed] 2002, Elsevier. 10. Laskaris G. Pocket atlas of oral [CrossRef] 3. Shear M, Speight P, Cysts of the diseases, 2006, Second edition, 17. Schulz MK, Teixeira LR, oral and maxillofacial regions, 4th ed., Thieme, Fernandes D, Bufalino A, Leon JE. Al- Blackwell Publishing Ltd 11. Moskow BS, Bloom A. Em- veolar cyst of the adult: Counterpart 4. Coulthard P, Horner K, Sloan P, bryogenesis of the gingival cyst. J of gingival cyst of the adult in eden- Theaker E, Oral and maxillofacial sur- Clin Periodontol. 1983 Mar;10(2): tulous ridge. J Oral Maxillofac Surg gery, radiology, pathology and oral 119–30. [PubMed] [CrossRef] Med Pathol. Available online 10 April medicine; First edition; 2003, Elsevier 12. Bhaskar SN, Laskin DM. 2018. [CrossRef] Science Gingival cysts; Report of three cases. 18. Wagner VP, Martins MD, Curra 5. Neville BW, Damm DD, Allen Oral Surg Oral Med Oral Pathol. 1955 M, Martins MA, Munerato MC. CM, Bouquot JE. Oral and Maxillofa- Aug;8(8):803-7. [PubMed] Gingival Cysts of Adults: Retrospec- cial Pathology. In: Neville BW, Damm 13. Sato H, Kobayashi W, Sakaki H, tive Analysis from Two Centers in DD, Allen CM, Bouquot JE, editors. Kimura H. Huge Gingival Cyst of the South Brazil and a Review of the Lit- Odontogenic cysts and tumors. 3rd ed. Adult: A Case Report and Review of erature. J Int Acad Periodontol. 2015 St. Louis: Saunders; 2009. p. 692. the Literature. Asian J Oral Jan;17(1): 14-9. [PubMed] 6. Marx R, Stern D, Oral and max- Maxillofac Surg. 2007 Sep;19(3):176- 19. Kelsey WP 5th, Kalmar JR, illofacial pathology - a rationale for di- 8. [CrossRef] Tatakis DN. Gingival cyst of the adult: agnosis and treatment, first edition, 14. Nxumalo TN, Shear M. regenerative therapy of associated root 2003,Quintessence Publishing Co Gingival cyst in adults. J Oral Pathol exposure. A case report and literature 7. Scully C, de Almeida O, Bagan Med. 1992 Aug;21(7):309-13. review. J Periodontol. 2009 Dec; J, Dios PD. Oral medicine and pathol- [PubMed] [CrossRef] 80(12):2073-81. [PubMed] [CrossRef] ogy at a glance; Wiley-Blackwell,

Please cite this article as: Deliverska EG, Stamatoski A. Gingival cyst of adults- two case reports and literature review. J of IMAB. 2018 Apr-Jun;24(2):2065-2068. DOI: https://doi.org/10.5272/jimab.2018242.2065

Received: 28/02/2018; Published online: 21/06/2018

Address for correspondence: Elitsa Georgieva Deliverska, Associate Professor, Department of Oral and Maxillofacial surgery, Faculty of Dental Medicine, Medical University Sofia, 1, Georgi Sofiiski Blvd., 1431 Sofia, Bulgaria. E-mail: [email protected] 2068 https://www.journal-imab-bg.org J of IMAB. 2018 Apr-Jun;24(2)