Journal of Indian Society of Periodontology
Journal of Indian Society of Periodontology
Home | About JISP | Search | Accepted articles | Online Early | Current Issue | Archives | Instructions | SubmissionSubscribeLogin 
Users Online: 443  Home Print this page Email this page Small font size Default font size Increase font sizeWide layoutNarrow layoutFull screen layout


 
   Table of Contents    
CASE REPORT
Year : 2018  |  Volume : 22  |  Issue : 6  |  Page : 546-550  

Recurrent gingival cyst of adult: A rare case report with review of literature


Department of Periodontology, Manipal College of Dental Sciences, Mangalore Manipal Academy of Higher Education, Manipal, Karnataka, India

Date of Submission05-Jun-2018
Date of Acceptance03-Aug-2018
Date of Web Publication1-Nov-2018

Correspondence Address:
Dr. Sangeeta Umesh Nayak
Department of Periodontology, Manipal College of Dental Sciences, Mangalore Manipal Academy of Higher Education, Manipal - 576 104, Karnataka
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jisp.jisp_382_18

Rights and Permissions
   Abstract 


The gingival cyst of adult (GCA) is a rare developmental anomaly of odontogenic origin. It is asymptomatic, slow growing, and commonly seen near the canine and premolar region of mandible. The GCA is usually treated by excisional biopsy. Usually, occurrence is more common in 5th to 6th decade of life. The present case report describes a solitary GCA treated by excisional biopsy in a 76-year-old male patient which had recurred for the fourth time. This article also highlights on diagnostic process and a review of literature describing its histiogenesis, clinical, and histopathologic features of this condition.

Keywords: Excisional biopsy, gingival cyst of adult, odontogenic cyst


How to cite this article:
Karmakar S, Nayak SU, Kamath DG. Recurrent gingival cyst of adult: A rare case report with review of literature. J Indian Soc Periodontol 2018;22:546-50

How to cite this URL:
Karmakar S, Nayak SU, Kamath DG. Recurrent gingival cyst of adult: A rare case report with review of literature. J Indian Soc Periodontol [serial online] 2018 [cited 2018 Dec 19];22:546-50. Available from: http://www.jisponline.com/text.asp?2018/22/6/546/244568




   Introduction Top


Gingival cyst of adults (GCAs) is a rare, noninflammatory, developmental cyst derived from the rests of dental lamina.[1],[2] The GCA typically occurs in the facial gingiva of the canine-premolar regions of mandible and is sometime confused with lateral periodontal cysts. They can also sometimes occur in the maxilla. Clinically, the lesion appears as a small, painless, flesh-colored swelling which may sometimes take up a bluish hue to the presence of cystic fluid.[3] It frequently occurs as a lone entity but may occur in multiples[1],[4] either unilaterally or bilaterally even on lingual aspect of the gingiva. GCA has been reported to cause a superficial saucerization on the alveolar bone surface due to pressure resorption of cystic contents. This resorption is seen during its excision. They are usually found in patients between 50 and 60 years of age.[1] Histopathologically the epithelial lining is seen as nonkeratinized with or without focal areas of thickening containing clear cells of glycogen-rich cytoplasm.[5]

GCA was first studied as an independent entity by Ritchie and Orban.[6] They termed it as gingival microcyst. However, their reports considered it to be just a histological curiosity without any clinical interest. The GCA was first reported as a distinct clinical entity with characteristic clinical and radiographic features by Bhaskar and Laskin.[7] Subsequent reports showed that the incidence to be 0.5%. Asians have a lower prevalence than Caucasians; however, there is no gender predilection.[3]

The following case reports describe a GCA with complete history, clinical, and histological manifestations as well as its treatment.


   Case Report Top


A 76-year-old male patient reported to the Department of Periodontology, complaining of a recurring swelling in the gum on the left lower front jaw for the past 3 months. History revealed that the swelling started gradually and increased to its present size. The patient does not report of any pain or discharge from the swelling. The patient every time used to visit a private dental clinic for treatment but repeatedly reported of recurrence within 2 weeks. The patient reported that this is the fourth time the swelling has recurred. He also does not recollect any history of trauma in this region. The patient stated that no microscopic examination of the sample was done earlier. Each time, the site and appearance of the swelling was very much similar.

Medical history of the patient revealed that he is a controlled hypertensive and is on aspirin 75 mg. The patient had earlier visited the dentist 20 years' ago for prosthetic rehabilitation. Extraoral examination of the patient did not reveal any palpable submental and submandibular lymph nodes.

Intraoral examination revealed a sessile, smooth, sharply demarcated swelling of bluish hue measuring approximately (5 mm × 5 mm × 5 mm), located in the attached gingiva with its lower border approximating the mucogingival junction on the facial side of 32 and 33. On palpation, the swelling was soft, had a cystic consistency, nontender, slightly movable, depressible with no discharge or bleeding. 31, 32, and 33 revealed a pocket depth of 2 mm with no bleeding on probing. 33 reveals Millers class I gingival recession. Pulp vitality testing of lateral incisor and canine revealed them to be vital. Radiographic analysis revealed no osseous anomaly [Figure 1] and [Figure 2].
Figure 1: Preoperative presentation of the lesion. (a) Labial view; (b) Mesial view; (c) Intraoral periapical with respect to 31, 32, and 33 showing no radiographic involvement

Click here to view
Figure 2: Dimensions of the cyst. (a) 5 mm mesiodistally; (b) 5 mm apicocoronally and (c) 5 mm in height

Click here to view


Based on clinical and radiographic manifestations, the provisional diagnosis of a cystic lesion of gingiva was made. The differential diagnosis included a lateral periodontal cyst, peripheral fibroma, parulis, odontogenic keratocyst, peripheral giant cell lesion, melanocytic nevus, angioleiomyoma, Kaposi sarcoma, and melanoma.

The diagnosis of lateral periodontal cyst was rejected as there was no radiographic finding suggesting of osseous involvement. Peripheral fibroma was ruled out because the lesion was soft and cystic. Parulis was ruled out because there was no periodontal pocket associated with the swelling and also probing revealed no pus discharge. As the lesion was not accompanied by any pain or bony expansion which was determined by its cystic consistency and absence of any radiographic changes the diagnosis of odontogenic keratocyst was not entertained.

Peripheral giant cell granuloma may have a bluish hue and smooth surface as seen in the present case. However, they are typically accompanied by superficial ulceration which is not seen in this case. It has also been observed to frequently involve the marginal gingiva which in the present case was absent.[8],[9] The oral melanocytic nevus can clinically present as a single bluish nodule; however, it is most commonly seen in hard palate and buccal mucosa with the involvement of attached gingiva just 12% of all reported cases.[10],[11] Angioleiomyomas can also give the appearance of a bluish painless nodule. However, they are uncommon in the oral cavity and affect mainly the lips and palate of middle-aged persons and rarely on the gingiva.[12],[13] Hence, its diagnosis was ruled out. Kaposi sarcoma can also present itself as well-delimited, small, bluish nodule on gingiva. But since, it is related to human immunodeficiency virus.[14],[15] infection its diagnosis was rejected. Oral melanoma usually occurs as dark bluish or black macule or nodule affecting the palate and gingiva of adult patients.[16] However, as the lesion progresses, it becomes irregular in shape accompanied by superficial ulceration, variable discoloration, and rapid growth which as it is not appreciated in this so this diagnosis was also ruled out. Thus, by exclusion, our diagnosis favored gingival cyst of adult.

Oral prophylaxis was performed. Since the patient was on aspirin therapy, a permission letter for informed consent for the excision of the lesion was written to his cardiologist, and oral hygiene instructions were given to the patient. After 3 days, the patient returned with the physician's consent and he was planned for excision of the lesion. Local anesthesia without adrenalin as the patient was hypertensive was administered by infiltration with respect to 32 and 33. An incision was placed with #11 blade at the base of the swelling. On incising a brown color fluid discharged. The lesion (cystic lining) was held with tweezers and was excised from its base by partial thickness incision. The surgical specimen Measuring approximately 6mmx6mmx6mm (1mm healthy surrounding tissue) was placed in 10% neutral-buffered formalin and sent to the Department of Oral Pathology for histological analysis [Figure 3].
Figure 3: Clinical view of surgical treatment. (a) Local anesthesia administered; (b) Incision made by #11 blade; (c) Postoperative view; (d) Periodontal pack placed

Click here to view


After the lesion was removed a (3 mm × 3 mm) defect in the attached gingiva was appreciated. On careful examination, it was verified underlying bone was not exposed. The area was irrigated with betadine and sterile saline. A periodontal pack was placed, postoperative instructions were given, and the patient was dismissed. The patient was prescribed 0.2% chlorhexidine gluconate two times per day for 2 weeks. The patient was recalled after 1 week [Figure 3].

Macroscopic examination revealed the specimen to be white to red, with a smooth surface, soft to firm in consistency measuring approximately (0.7 cm × 0.5 cm × 0.3 cm). The microscopic picture reveals the presence of mucosal surface lined by stratified squamous epithelium supported by a fibrous connective tissue containing a cystic space. The cystic lumen is lined by 2–3 cell thick flat to cuboidal cells supported by a fibrous connective tissue capsule exhibiting mild chronic inflammatory cell infiltrate chiefly lymphocytes and engorged blood vessels. The histopathologic diagnosis is gingival cyst of adult which confirms our clinical diagnosis [Figure 4].
Figure 4: Histopathology. (a) Low-power view. Mucosal surface (black arrows) lined by stratified squamous epithelium supported by a fibrous connective tissue containing a cystic space; (b) High-power view. The cystic lumen is lined by 2–3 cell thick flat to cuboidal cells (black arrows) supported by a fibrous connective tissue capsule exhibiting mild chronic inflammatory cell infiltrate chiefly lymphocytes and engorged blood vessels

Click here to view


After 1 week, the periodontal pack was removed. Complete reepithelialization of the surgical site was appreciated. The patient follow-up was done again at 3 and 6 months. Mild keratinization of the surgical site was observed initially and later complete healing as appreciated. No recurrence of the lesion was noted, and a definite mucogingival junction had formed [Figure 5].
Figure 5: Postoperative view. (a) Clinical view after 1 week; (b) Clinical view after 6 month (facial view); (c) Clinical view after 6 month (mesial view)

Click here to view



   Discussion Top


The present case report describes a case of gingival cyst of adult successfully treated with excisional biopsy.

Our review of literature has identified 177 reported cases of gingival cyst of an adult.[1],[3],[17] Studies stated that the mean age in which GCA has been observed is 51 years.[4] with a slight female predilection.[1] In this case, the patient was a 76-year-old male. Normally, the gingival cyst appears round-to-oval, firm in consistency, and located either in attached gingiva, alveolar mucosa or both. 76% of GCAs occur as a single lesion on the buccal aspect of the mandibular canine premolar area but may occur lingually.[1],[18] GCAs can also occur bilaterally.[1],[4] Although botryoid variant[1],[5],[19] and multiple cysts[1],[20] but are infrequent and just represent 24% of all reported cases. The size of the GCAs range from ~5 mm to 5 cm.[3] In the present case, it was round in shape, cystic in consistency and measuring (5 mm × 5 mm) and was located in the facial attached gingiva with respect to lateral incisor and canine. The swellings sometimes exhibit a bluish hue throughout their convexity due to the presence of cystic contents, but usually, they show little or no color change. The lesion, in this case, was bluish in color.

There was no radiographic involvement in this case. Only 22.5% of all reported cases have shown radiographic findings. Usually, a radiograph cannot accurately show the contour of this soft-tissue originating lesion as the cyst may involve either the buccal or lingual alveolar surface with a shallow “saucer-like” defect described first by Bhaskar and Laskin.[7] Only a single case reports of accompanying root exposure.[1] However, it is seen that at the time of cyst excision the osseous involvement is 45% clearly indicating the inability of radiographic to show definitive findings.

Different theories have been put forward to explain the histogenesis of GCA: (1) a differentiation from heterotopic salivary glandular tissue.[1],[6] (2) cystic degeneration of epithelial fragments implanted into the connective gingival tissue after trauma; and (3) development of GCA from the remnants of dental lamina: (rests of Serres) more frequently present on the vestibular side of the keratinized gingiva[2],[6],[19] or cell rests of Malassez.[1] The hypothesis that the GCA is an odontogenic derivative of dental lamina is based on human fetal head studies.[1] which discovered that the stalk of epithelium connecting the developing tooth and the dental lamina disintegrates as early as nine to 10 weeks in utero and leaves behind epithelial rests which come to be known as glands of Serres. This epithelium stalk is predisposed to cystic disintegration as seen by its tendency to degrade even before the dental lamina has broken down.[2] As a result of this, the microcysts are exfoliated into the oral cavity during tooth eruption where some may endure into adulthood and come to be known as GCA.[1]

Excisional biopsy is the treatment of choice for GCA. 100% of the reviewed cases have reported to be treated by this treatment modality. The technique involves making an incision at the base of the lesion and then extending it along the lesion's circumference. After this enucleation is carried out. In all the cases except four[4],[17],[21] the surgical sites were allowed to heal by secondary intention. In one case, the soft-tissue defect was treated by free autogenous gingival graft.[22] In another case,[1] as there was osseous involvement, the defect was filled by freeze-dried bone allograft, and it was covered by bovine tendon collagen membrane. These were held secure by sutures.

Along with this case report, 27 cases[1],[5],[18],[20],[21],[23],[24] were followed up, and none reported the recurrence of the lesion. Among these, only 22[1],[5],[18],[20] including this present case report gave a specific follow -p period which ranged from ranged from 1 month to 3 years, with the average time being 1.2 years. Radiographic follow-up was given by only two case reports.[1],[25] Both of them showed a complete resolution of radiographic findings.

The histopathologic findings of GCA are variable. Usually, the cystic lining is made up of either nonkeratinized squamous or cuboidal epithelium, with a thickness ranging from one to five cell layers thick. Sometimes, clear cells with glycogen-rich cytoplasm are noted.[18],[22] Cystic lining comprising of even respiratory epithelium.[26] A case reports of pigmented melanocytes dispersed throughout the epithelial layer.[1] Although some cases report the cystic lumen to contain keratin or amorphous eosinophilic material[1],[5],[10],[23] and calcified material,[2] it is mostly empty.


   Conclusion Top


The GCA is a unique developmental cyst of odontogenic origin. It is routinely seen in the mandibular canine premolar area in adults in 50–60 years of age. The alveolar process is less frequently involved and is usually not detected in radiographs. GCA is routinely treated with excisional biopsy. Early identification of the lesion and histopathological examination helps in providing prompt treatment. This needs a multidisciplinary approach and management.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Kelsey WP 5th, Kalmar JR, Tatakis DN. Gingival cyst of the adult: Regenerative therapy of associated root exposure. A case report and literature review. J Periodontol 2009;80:2073-81.  Back to cited text no. 1
    
2.
Moskow BS, Bloom A. Embryogenesis of the gingival cyst. J Clin Periodontol 1983;10:119-30.  Back to cited text no. 2
    
3.
Sato H, Kobayashi W, Sakaki H, Kimura H. Huge gingival cyst of the adult: A case report and review of the literature. Asian J Oral Maxillofac Surg 2007;19:176-8.  Back to cited text no. 3
    
4.
Giunta JL. Gingival cysts in the adult. J Periodontol 2002;73:827-31.  Back to cited text no. 4
    
5.
Hegde U, Reddy R. Gingival cyst of adult – A case report with unusual findings. Indian J Dent Res 2004;15:78-80.  Back to cited text no. 5
    
6.
Ritchey B, Orban B. Cysts of the gingiva. Oral Surg Oral Med Oral Pathol 1953;6:765-71.  Back to cited text no. 6
    
7.
Bhaskar SN, Laskin DM. Gingival cysts; report of three cases. Oral Surg Oral Med Oral Pathol 1955;8:803-7.  Back to cited text no. 7
    
8.
Lester SR, Cordell KG, Rosebush MS, Palaiologou AA, Maney P. Peripheral giant cell granulomas: A series of 279 cases. Oral Surg Oral Med Oral Pathol Oral Radiol 2014;118:475-82.  Back to cited text no. 8
    
9.
Katsikeris N, Kakarantza-Angelopoulou E, Angelopoulos AP. Peripheral giant cell granuloma. Clinicopathologic study of 224 new cases and review of 956 reported cases. Int J Oral Maxillofac Surg 1988;17:94-9.  Back to cited text no. 9
    
10.
Buchner A, Hansen LS. Pigmented nevi of the oral mucosa: A clinicopathologic study of 36 new cases and review of 155 cases from the literature. Part II: Analysis of 191 cases. Oral Surg Oral Med Oral Pathol 1987;63:676-82.  Back to cited text no. 10
    
11.
Meleti M, Mooi WJ, Casparie MK, van der Waal I. Melanocytic nevi of the oral mucosa-no evidence of increased risk for oral malignant melanoma: An analysis of 119 cases. Oral Oncol 2007;43:976-81.  Back to cited text no. 11
    
12.
Gueiros LA, Romañach MJ, Pires-Soubhia AM, Pires FR, Paes-de-Almeida O, Vargas PA. Angioleiomyoma affecting the lips: Report of 3 cases and review of the literature. Med Oral Patol Oral Cir Bucal 2011;16:e482-7.  Back to cited text no. 12
    
13.
Brooks JK, Nikitakis NG, Goodman NJ, Levy BA. Clinicopathologic characterization of oral angioleiomyomas. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2002;94:221-7.  Back to cited text no. 13
    
14.
Rockafellow A, Ko E, Philipone E. Asymptomatic gingival lesion occurring in an 83-year-old man. Oral Surg Oral Med Oral Pathol Oral Radiol 2014;118:5-8.  Back to cited text no. 14
    
15.
Lager I, Altini M, Coleman H, Ali H. Oral Kaposi's sarcoma: A clinicopathologic study from South Africa. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2003;96:701-10.  Back to cited text no. 15
    
16.
Sortino-Rachou AM, Cancela Mde C, Voti L, Curado MP. Primary oral melanoma: Population-based incidence. Oral Oncol 2009;45:254-8.  Back to cited text no. 16
    
17.
Gil Escalante M, Tatakis DN. Gingival cyst of the adult as early sequela of connective tissue grafting. Case Rep Dent 2015;2015:473689.  Back to cited text no. 17
    
18.
Wysocki GP, Brannon RB, Gardner DG, Sapp P. Histogenesis of the lateral periodontal cyst and the gingival cyst of the adult. Oral Surg Oral Med Oral Pathol 1980;50:327-34.  Back to cited text no. 18
    
19.
Nxumalo TN, Shear M. Gingival cyst in adults. J Oral Pathol Med 1992;21:309-13.  Back to cited text no. 19
    
20.
Wescott WB, Correll RW, Craig RM. Two fluid-filled gingival lesions in the mandibular canine-first premolar area. J Am Dent Assoc 1984;108:653-4.  Back to cited text no. 20
    
21.
Brod JM, Passador-Santos F, Soares AB, Sperandio M. Gingival cyst of the adult: Report of an inconspicuous lesion associated with multiple agenesis. Case Rep Dent 2017;2017:4346130.  Back to cited text no. 21
    
22.
Dent CD, Rubis EJ, MacFarland PJ. Bilateral gingival swellings in the mandibular canine-premolar areas. J Am Dent Assoc 1990;120:71-2.  Back to cited text no. 22
    
23.
Fardal O, Johannessen AC. Rare case of keratin-producing multiple gingival cysts. Oral Surg Oral Med Oral Pathol 1994;77:498-500.  Back to cited text no. 23
    
24.
Malali VV, Satisha TS, Jha AK, Rath SK. Gingival cyst of adult: A rare case. J Indian Soc Periodontol 2012;16:465-8.  Back to cited text no. 24
[PUBMED]  [Full text]  
25.
Holder TD, Kunkel PW Jr. Case report of a periodontal cyst. Oral Surg Oral Med Oral Pathol 1958;11:150-4.  Back to cited text no. 25
    
26.
Young LL, Reeve CM, Frantzis TG. Gingival cyst lined with respiratory epithelium: Report of a case. J Periodontol 1972;43:490-1.  Back to cited text no. 26
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]



 

Top
   
 
  Search
 
  
    Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
    Access Statistics
    Email Alert *
    Add to My List *
* Registration required (free)  

 
  In this article
    Abstract
   Introduction
   Case Report
   Discussion
   Conclusion
    References
    Article Figures

 Article Access Statistics
    Viewed147    
    Printed2    
    Emailed0    
    PDF Downloaded52    
    Comments [Add]    

Recommend this journal