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Table of Contents
CASE REPORT
Year : 2022  |  Volume : 13  |  Issue : 4  |  Page : 121-126  

Chondroid choristoma of the tongue: A rare case report


1 Department of Dentistry, Pediatric and Preventive Dentistry, Nalanda Medical College Hospital, Sadikpur, Bihar, India
2 Department of Dentistry, Oral Pathology and Microbiology, Nalanda Medical College Hospital Agamkuan Flyover, Sadikpur, Bihar, India
3 Prosthodontics and Crown and Amp, National Institute of Medical Sciences, Jaipur, Rajasthan, India
4 Department of Pediatric and Preventive Dentistry, Buddha Institute of Dental Sciences and Hospital, Kankadbagh, Bihar, India

Date of Submission10-Dec-2019
Date of Acceptance20-Apr-2020
Date of Web Publication26-May-2022

Correspondence Address:
Dr. Shabab Ahmed Khan
Prosthodontics and Crown and Amp, National Institute of Medical Sciences, Jaipur, Rajasthan
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/njms.NJMS_93_19

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   Abstract 


Choristomas are tumor-like masses consisting of normal cells in an abnormal location. Choristomas of the oral cavity are rare lesions. We report a case of Cartilaginous choristoma on the ventral aspect of the tongue in a 25-year-old female. Clinical features, differential diagnosis, and tumoral origin theories are also discussed along with a meta-analysis of the reported cases in the PubMed database.

Keywords: Benign, cartilage, chondroid choristoma, tongue


How to cite this article:
Nezam S, Nishat R, Khan SA, Shukla JN. Chondroid choristoma of the tongue: A rare case report. Natl J Maxillofac Surg 2022;13, Suppl S1:121-6

How to cite this URL:
Nezam S, Nishat R, Khan SA, Shukla JN. Chondroid choristoma of the tongue: A rare case report. Natl J Maxillofac Surg [serial online] 2022 [cited 2022 Oct 5];13, Suppl S1:121-6. Available from: https://www.njms.in/text.asp?2022/13/4/121/346017




   Introduction Top


The term “Choristoma” can be used to describe tumor-like masses consisting of normal cells in an abnormal location, i.e., a “heterotopic” rest of the cells.[1],[2] The occurrence of these entities has been attributed to abnormalities in the embryonic development of the neural tube.[2] It is crucial to distinguish choristomas from hamartomas and teratomas, wherein hamartomas are nonneoplastic, unifocal/multifocal, and developmental malformations, comprising a mixture of cytologically normal mature cells and tissues which are indigenous to the anatomic location, showing disorganized architectural pattern with predominance of one of its components.[3] On the other hand, teratomas are true neoplasms composed of a variety of parenchymal cell types representative of more than one germ layer.[4] Choristomas found in the oral cavity are classified according to the type of tissue, including salivary gland, Cartilaginous, osseous, thyroid, sebaceous, glial, and gastric/respiratory mucosal.[2]

Cartilaginous choristomas in the oral cavity are unusual entities. We present one such rare case of Cartilaginous choristoma on the ventral aspect of the tongue.


   Case Report Top


A 28-year-old female presented with an asymptomatic mass on the ventral aspect of the tongue of 2-year duration. The patient did not give any history of trauma or any other lesion in the oral cavity. The lesion had gradually increased in size in the past 2 years. On intraoral examination, a firm, nontender, well-demarcated, multilobulated submucosal mass measuring approximately 2.3 cm × 1.6 cm was seen on the left ventral aspect of the tongue crossing the midline [Figure 1]. The overlying mucosa was not associated with any inflammation or ulceration but showed areas of whitish discoloration. Tongue movements were normal. A provisional diagnosis of a benign mesenchymal neoplasm was given. Differential diagnosis included salivary gland neoplasm and granular cell tumor. Excisional biopsy of the lesion was done under local anesthesia, and the excised specimen was sent for histopathological evaluation. The gross specimen measured around 2.6 cm × 1.9 cm, was roughly oval in shape, firm in consistency, and reddish-white in color, and had a lobulated surface. Histopathological examination revealed a nonkeratinized stratified squamous epithelium and an underlying fibrovascular stroma. Deeper part of the stroma showed lobular proliferation of basophilic mature hyaline cartilage surrounded by a fibrotic capsule, with typical chondroblasts arranged in cell nests or in isolation [Figure 2] and [Figure 3]. No atypia was evident. A final diagnosis of chondroid choristoma was given. No evidence of recurrence was found in a 6-month follow-up [Figure 4].
Figure 1: Clinical photograph showing a firm, nontender, well-demarcated, multilobulated submucosal mass on the right ventral aspect of the tongue crossing the midline

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Figure 2: Photomicrograph showing lobular cartilage proliferation (green arrow), fibrotic capsule (blue arrow)

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Figure 3: Photomicrograph showing chondrocytes in the lacunae (black arrows)

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Figure 4: Six months of follow-up

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   Discussion Top


Choristomas are lesions characterized by the presence of cells not native to the site. Oral choristomas can be composed of various types of tissues, including cartilage, bone, salivary gland, thyroid, sebaceous, glial, respiratory, and gastrointestinal tissue.[2] Oral Cartilaginous choristomas are rare entities with only 32 cases being reported till date, ours being the 33rd case [[Table 1] provides a meta-analysis of all the reported cases in the PubMed literature].
Table 1: Cases of chondroid choristomas of tongue reported in the English literature since 1890

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These have been reported in patients ranging between 3 and 80 years of age, with tongue being the most common site of occurrence followed by gingiva, buccal mucosa, soft palate, and palatine tonsil. Sixty percent of the tongue lesions are seen on the dorsum of tongue, 32% on the lateral border, and only 8% on the ventral surface.[25],[31],[32]

The term choristoma in the oral cavity was introduced in 1971 by Knoll et al.[33] Zegrelli et al. stated that the term “Cartilaginous choristoma of the tongue” should be used to describe only those lesions that show exclusive chondromatous growth.[12] Several theories have been put forward to explain the origin of cartilage in the oral soft tissue; however, the exact etiology of the lesion still remains unclear. According to the embryonal theory, cartilage develops from the heterotropic fetal Cartilaginous remnants.[34] Remnants of Meckel's cartilage or displacement of Cartilaginous elements from the first four branchial arches to the area of tongue may act as possible sources for the entity to develop.[2] Metaplastic theory states that trauma, irritation, or chronic inflammation could stimulate the pluripotent mesenchymal cells to differentiate into chondrocytes and proliferate to form the lesion, or this transformation could occur de novo.[31],[35],[36] Chou et al. postulated that proper stimulation and active interstitial and appositional growth of multipotent mesenchymal cells could result in the cartilage formation.[37] It was also hypothesized that the vestigial rests of cartilage could act as a source of origin of chondroid choristomas.[2],[11] Chromosomal abnormalities involving the 12q13–q15 region have been associated with chondroid choristomas of the soft tissues.[38]

Differential diagnosis of chondroid choristoma includes a variety of benign lesions such as chondroma, pleomorphic adenoma, traumatic chondromatous metaplasia, ectomesenchymal chondromyxoid tumor, and granular cell tumor. It is also important to distinguish it from malignant Cartilaginous neoplasms such as primary chondrosarcoma or metastasis from a primary intraosseous chondrosarcoma.[28],[30],[31]

In the present case, the absence of epithelial and mesenchymal components, such as plasmacytoid cells, fusiform cells, cuboidal cells, chondromyxoid stroma along with the absence of morphological patterns of the epithelial cells (trabecular, ductal, cystic, and solid) helped in differentiating it from pleomorphic adenoma.[39] Absence of lobulated growth pattern, clusters of chondrocytic cells, and surrounding collagenous stroma excluded the diagnosis of chondroma.[30],[40] Traumatic chondromatous metaplasia is seen in edentulous ridges as a result of chronic mechanical irritation from ill-fitting dentures.[1] In our case, the patient was dentulous, and hence, this differential diagnosis was ruled out. Ectomesenchymal chondromyxoid tumor is histopathologically characterized by the lobular proliferation of ovoid and fusiform cells with occasional foci of atypia in a chondromyxoid background and absence of these features eliminated this diagnosis.[41] Absence of microscopic features such as irregular nuclear morphology, aberrant intercellular matrix formation, increased cellularity, mitoses, and invasion into surrounding tissues ruled out the malignant Cartilaginous neoplasms.[24] Granular cell tumor was ruled out due to the absence of accumulation of plump cells with abundant granular cytoplasm.[42]

Choristomas of various histological subtypes and in different anatomical locations of the oral cavity have been reported. Chondroid choristomas of the palatine tonsils have been reported by Thada et al., Bharti et al., and Bedi et al.[43],[44],[45] Malis et al. reported a case of chondroid choristoma in the nasopharynx.[46] Saha et al. reported a case of chondroid choristoma in the maxillary labial vestibule in an 11-year-old girl which was present since birth.[47] A rare case of glial choristoma of the tongue associated with cleft palate was reported in a 3-month-old male infant by Yamada et al.[48] Suganya et al. reported a case of Cartilaginous choristoma of the gingiva.[1] Jorquera et al. reported a rare case of lingual choristoma lined by respiratory and gastric epithelium.[49] Osseous choristomas of the oral cavity have been reported by Bastian et al. and Adhikari et al.[50],[51],[52] A rare case report of an osseous choristoma was reported by Karci et al. in the periodontium.[52] Epidermal choristomas of the oral cavity have been reported by Chi et al.[53]

Analysis of chondroid choristomas occurring on the tongue in the published PubMed literature and the present case showed a total of 33 cases with 17 cases occurring in females and 16 in males, thus showing a slight female predilection. The lesion was seen in a wide age range, ranging between 5 and 75 years of age with a mean age of 38.3 years. Patients gave a history of the lesion being present for duration of few months to 30 years. Most of the lesions occurred on the dorsum of the tongue followed by lateral border and then by the ventral aspect of the tongue. Excision of the lesion was done in all the cases, and none of the authors reported any evidence of recurrence. One case co-existed with squamous cell carcinoma, in which hemiglossectomy with radical neck dissection was done [Table 1].

In conclusion, chondroid choristomas are benign developmental lesions which need to be diagnosed correctly for apt management. Free marginal surgical excision is the best treatment of choice. No recurrences have been reported in the literature.

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 initials 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.



 
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