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An Official Publication of the Indian Association of Oral and Maxillofacial Pathologists


 
  Table of Contents    
CASE REPORT  
Year : 2021  |  Volume : 25  |  Issue : 4  |  Page : 90-93
 

Gingival salivary gland choristoma: An unusual case report


1 Department of Microbiology and Pathology, Pathology Research and Diagnosis Center; Department of Dental Clinics, Oral Medicine Clinic, University Center of Health Sciences, University of Guadalajara, Guadalajara, Jalisco, México
2 Department of Microbiology and Pathology, Pathology Research and Diagnosis Center, University Center of Health Sciences, University of Guadalajara, Guadalajara, Jalisco, México

Date of Submission24-Sep-2019
Date of Decision05-Jan-2021
Date of Acceptance19-Jan-2021
Date of Web Publication19-Mar-2021

Correspondence Address:
José Sergio Zepeda Nuño
Department of Microbiology and Pathology, University Center of Health Sciences, University of Guadalajara, Sierra Mojada #950, Colonia Independencia, Guadalajara, Jalisco, C.P. 44340
México
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jomfp.JOMFP_284_19

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   Abstract 


The gingival salivary gland choristoma, a highly unusual tumor-like mass alteration constituted by normal salivary gland cells in an abnormal location, reported for the first time in 1964 by Moskow and Baden. To our knowledge, only 12 cases of this entity (including present case) are reported to date, the majority observed as asymptomatic, solitary, smooth-surfaced tumor-like masses, measuring 0.5–1.5 cm with no osseous involvement. This case involves a 38-year-old female, with a pink symptomatic polypoid nodule on the posterior mandibular alveolar ridge mucosa, measuring 6 mm in diameter where no osseous abnormalities are shown. An excisional biopsy was performed. Microscopically, the specimen was constituted by dense fibrous connective tissue containing mucous minor salivary glands, intraductal calcification and adipose tissue clusters. Furthermore, inflammatory infiltrate foci were seen. An immunohistochemical technique was used as the support for the diagnostic methodology. The diagnosis of gingival salivary gland choristoma was established. Some development theories are discussed, referring to a pluripotential capacity of the gingiva. This case reflects the importance of not underestimating innocuous lesions that could represent more serious or unusual entities. In addition, histopathological analysis is mandatory to achieve a correct diagnosis and management of soft-tissue enlargements of oral mucosa.


Keywords: Alveolar ridge, gingival choristoma, heterotopic tissue, polypoid nodule, salivary gland


How to cite this article:
Torres Medina MH, Topete RF, Villalba MN, Zepeda Nuño JS. Gingival salivary gland choristoma: An unusual case report. J Oral Maxillofac Pathol 2021;25:90-3

How to cite this URL:
Torres Medina MH, Topete RF, Villalba MN, Zepeda Nuño JS. Gingival salivary gland choristoma: An unusual case report. J Oral Maxillofac Pathol [serial online] 2021 [cited 2021 Apr 11];25:90-3. Available from: https://www.jomfp.in/text.asp?2021/25/4/90/311547





   Introduction Top


The gingival salivary gland choristoma (GSGC) is a very unusual salivary gland alteration, described for the first time in 1964 by Moskow and Baden.[1] The term choristoma is defined as a cohesive tumor-like mass constituted by normal cells in an abnormal location.[2],[3] To our knowledge, only 12 well-documented cases of GSGC (including present case) are reported to date [Table 1].[1],[4],[5],[6],[7],[8],[9],[10],[11],[12] Other similar alterations have been reported and classified as gingival cyst derived from the ectopic salivary gland tissue. Although these entities present variations from the original case, the majority of GSGC are reported as asymptomatic, solitary, smooth-surfaced tumor-like masses, measuring 0.5–1.5 cm in diameter where no osseous involvement was observed.[2] In order to support for the diagnostic methodology, an immunohistochemical technique was applied. The purpose of this case report is to present the discovery of an unusual alteration of salivary gland development where it is liable to give an erroneous diagnosis, due to the clinical appearance and the location of this entity. In addition, the possible origin of the pathology is discussed.
Table 1: Gingival salivary gland choristomas reported to date

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   Case Report Top


A 38-year-old female, was referred to the oral medicine service at the University of Guadalajara. At the diagnosis interrogation, the patient reported a 3-month symptomatic enlargement and did not mention any antecedents of importance. After obtaining consent from the patient, we proceed to intraoral exploration that revealed a polypoid nodule of approximately 6 mm in size, with smooth surface, firm consistency and similar coloration to the normal adjacent oral mucosa, the lesion was located on the posterior alveolar ridge mucosa close upon a root remnant of the mandibular right second molar [Figure 1]a. A periapical radiography was applied, where no osseous or dental root abnormalities are shown [Figure 1]b. An excisional biopsy was performed under local anesthesia, and the obtained tissue was submitted to the histopathological analysis.
Figure 1: Clinical and radiographic images. (a) A pedunculated swelling on the mandibular region close to root remnant of the second molar. (b) A periapical radiography where no bone involvement was noted

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Microscopically, the specimen was constituted by a nodular mass of dense fibrous connective tissue [Figure 2], containing mucous minor salivary glands [Figure 3]a, medium caliber arteries and lymphatic vessels. Adjacent to salivary acini, intraductal calcification [Figure 3]b and adipose tissue clusters [Figure 3]c could be observed. In the overall stroma, mild inflammatory infiltrate foci with predominance of lymphocytes were observed.
Figure 2: Photomicrography of the excisional specimen. Low-power view of the nodule, which is composed of connective and salivary gland tissue, the proliferation is lined by a keratinized stratified squamous epithelium (H and E, ×7)

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Figure 3: Representative photomicrographs of the excisional specimen. (a) Between the stroma, multiple mucous cells are observed in the salivary gland lobes, also intralobular ducts, vascular spaces and lymphatic capillaries (H and E, ×80). (b) Next to the salivary gland lobes, intraductal calcifications are shown (H and E, ×80). (c) Presence of mature adipose tissue (H and E, ×80)

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Although it is not necessary to establish the diagnosis, an immunohistochemical technique was performed to assess the proliferative activity of gland cells through the evaluation of Ki-67 expression, where it was observed that the majority of glandular cells show lack of expression [Figure 4]. In addition, positive expression of Ki-67 was found in the 2% and 4% of the nuclei of acinar and stromal cells, respectively. Based on the clinical and histopathological findings, the diagnosis of GSGC known as heterotopic salivary gland tissue was established. After the excisional biopsy, postsurgical care was indicated to the patient. The relief of occasional pain was achieved, and there was no recurrence after 12 weeks of follow-up.
Figure 4: Immunohistochemistry of Ki-67 staining (×400). Expression of Ki-67 in some stromal and acinar cell nuclei was observed (Ki-67, ×400). Inset: Internal positive control of the basal cell layer (×400)

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   Discussion Top


According to the clinical and radiographic findings, this injury can be diagnosed with a reactive origin tumor-like mass due to the close relationship of the alveolar ridge with the occlusal contact, similar to an irritation fibroma, which usually is a well-delimited smooth surface where a firm consistency is observed on the nodule. This can vary from millimeters to a few centimeters and the growth is attached to the mucosa by a sessile or pedunculated base, and mild symptomatology can be associated due to constant trauma.[13]

According to the data, it is uncertain how minor salivary glands cells have interacted to create mucinous acini in the mandibular alveolar ridge. Researches explain some theories about the mechanism of the development of GSGC. One of these theories describes that the gingival epithelium shows a pluripotential quality, so the unusual location of this minor salivary gland tissue demonstrates that there is an ectopic formation, thus creating a morphogenesis of minor salivary glands.[8] Therefore, the fact of finding adipose tissue in the present case reinforces the possible theory of the pluripotential capacity of the gingiva. Another theory debates that in the normal salivary gland tissue development, a fraction of glandular tissue placed in the oral mucosa becomes “trapped” in the gingiva and consequently producing an ectopic growth, leading to the formation of GSGC.[2]

In general, it is known that the choristoma is a mature tissue growth with no significant mitotic activity. In the present case, we found positive Ki-67 in some acinar cells. There are a few studies that evaluate index proliferation in adult salivary glandular tissue. In one study, Ki-67 positive cells were expressed in acinar and ductal cells, with a frequency of 8% and 1%, respectively.[14] In a more recent work, Aure et al. evaluated the proliferative capacity of the salivary glands in murine mice. They found that homeostasis and maintenance of the adult salivary gland cells are through the duplication of differentiated secretory cells.[15]


   Conclusion Top


The gingival mucosa is constantly under chronic irritation, chewing forces, trapped food remains, poorly-adjusted restorations, dental calculus, as well as the oral microbiota which under atypical conditions may become pathogenic. That is why in gingiva, there is a tendency for multiple entities to appear which are associated with chronic trauma and external irritation and could be a factor to present symptomatology. It is important that the professional of oral care do not underestimate the innocuous lesions, even polypoid nodules could represent more serious or unusual entities. Whereby a histopathological analysis is mandatory to achieve a correct diagnosis and management of soft-tissue enlargements of oral mucosa.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient (s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initial s will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Moskow BS, Baden E. Gingival choristoma. Report of a case. Oral Surg Oral Med Oral Pathol 1964;18:504-16.  Back to cited text no. 1
    
2.
Chou LS, Hansen LS, Daniels TE. Choristomas of the oral cavity: A review. Oral Surg Oral Med Oral Pathol 1991;72:584-93.  Back to cited text no. 2
    
3.
Batra R. The pathogenesis of oral choristomas. J Oral Maxillofac Surg Med Pathol 2012;24:110-4.  Back to cited text no. 3
    
4.
Traeger KA. Cyst of the gingiva (mucocele). Oral Surg Oral Med Oral Pathol 1961;14:243-5.  Back to cited text no. 4
    
5.
Moss-Salentijn L, Applebaum E. A minor salivary gland in human gingiva. Arch Oral Biol 1972;17:1373-4.  Back to cited text no. 5
    
6.
Wilson DF, MacEntee MI. Papillary cystadenoma of ectopic minor salivary gland origin. Oral Surg Oral Med Oral Pathol 1974;37:915-8.  Back to cited text no. 6
    
7.
Izumi H, Oikawa T, Yoshida T, Inoue Y, Komiyama K, Ryo T, et al. A consideration of a case of gingival cyst with glandular epithelium. Nihon Univ J Oral Sci 1976;2:242-6.  Back to cited text no. 7
    
8.
Ide F, Shimura H, Saito I, Umemura S. Gingival salivary gland choristoma: An extremely rare phenomenon. Oral Surg Oral Med Oral Pathol 1983;55:169-72.  Back to cited text no. 8
    
9.
Moskow BS, Baden E. Gingival salivary gland. Report of a case. J Clin Periodontol 1986;13:720-4.  Back to cited text no. 9
    
10.
Brannon RB, Houston GD, Wampler HW. Gingival salivary gland choristoma. Oral Surg Oral Med Oral Pathol 1986;61:185-8.  Back to cited text no. 10
    
11.
Ledesma-Montes C, Fernandez-Lopez R, Garces-Ortiz M, Portilla-Robertson J, Hernandez-Guerrero JC. Gingival salivary gland choristoma. A case report. J Periodontol 1998;69:1164-6.  Back to cited text no. 11
    
12.
Gheena S, Chandrasekhar T, Ramani P. Heterotopic salivary gland tissue: A report of two cases. J Nat Sci Biol Med 2011;2:125-7.  Back to cited text no. 12
    
13.
Neville BW, Damm DD, Allen CM, Chi AC. Soft tissue tumors. In: Neville BW, Damm DD, Allen CM, Chi AC, editors. Oral and Maxillofacial Pathology. 4th ed. St. Louis, Missouri: Elsevier; 2016. p. 473-97.  Back to cited text no. 13
    
14.
Murakami M, Ohtani I, Hojo H, Wakasa H. Immunohistochemical evaluation with Ki-67: An application to salivary gland tumours. J Laryngol Otol 1992;106:35-8.  Back to cited text no. 14
    
15.
Aure MH, Konieczny SF, Ovitt CE. Salivary gland homeostasis is maintained through acinar cell self-duplication. Dev Cell 2015;33:231-7.  Back to cited text no. 15
    


    Figures

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

  [Table 1]



 

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