| Abstract|| |
Rationale: Granular cell tumours (GCTs) of the tongue are a rare, soft tissue pathological entity at young ages. This case report aims to present one such case. Patient Concerns: A 16-year-old female patient sought treatment for a small, slow-growing, painless nodule in the dorsum of the tongue since six months. She underwent successful orthodontic treatment in recent past and is in the retention phase. Diagnosis: Excisional biopsy revealed the lesion to be a GCT under histopathology. Treatment: The patient was treated for an abnormal, small, slow-growing, painless nodule in the dorsum of the tongue. Outcomes: The patient had satisfactory esthetics and early diagnosis. The active intervention dispelled the confusion about the role of trauma and orthodontics appliance in the etiology of GCT in this particular case to the patient. Take-away Lessons: GCT can occur at any age. Early diagnosis and corrective surgery would help to avoid late complications.
Keywords: Granular cell tumour, oral reactive lesions, soft tissue lesion
|How to cite this article:|
Balaji S M, Balaji P. Rare case of granular cell tumour at 16 years – A case report. Indian J Dent Res 2022;33:216-8
| Background|| |
Granular cell tumour (GCT) is a rare benign neoplasm originating from neural Schwann cells. It presents often as single, insidious in origin, painless, slow-growing, well-delineated lesion in the fourth to sixth decade of life. Half of all reported cases occur in the head and neck region, with the tongue being the most favoured site. About 2% of cases exhibited malignant features. Different treatment approaches from regular monitoring to radical excision have been suggested., The origin of GCT is debated with a few of the reports indicating association with a traumatic episode., This manuscript aims to present a case of GCT in a 16-year old female patient who had completed orthodontic treatment in the immediate past.
| Case Report|| |
A 16-year-old female patient complained of a small, nodular swelling along the midline of the tongue for about six months. The swelling was insidious in origin, painless, and slow-growing. The patient attributed her past orthodontic treatment to the growth. The patient had no relevant family or personal history or relevant medical habits. The patient had completed orthodontic treatment about six months earlier for class-1 malocclusion with a rotated tooth that was treated with extraction of all four premolars. Currently, she is in the retention phase with lingual bonded retainers. The intraoral examination revealed a firm, well-circumscribed whitish lesion of about 1.0 cm in diameter, located along the midline at the middle of the tongue [Figure 1]. A standard excisional biopsy was performed, and appropriate sutures were placed. The patient was placed under ibuprofen 600 mg 8 hourly and antibiotic coverage for 5 days. The patient remained disease-free for six months and is under regular follow-up.
Histopathological study revealed it to be a submucosal lesion under a normal glossal epithelium and lesion made up of proliferating polygonal cells with a large clarified granular cytoplasm with a central, oval, and monotonous nucleus arranged in sheets, suggestive of GCT [Figure 2] and [Figure 3].
|Figure 2: (a): Scanner view of the lesion. Note the tongue epithelium and the non-encapsulated lesion-normal tissue interface. (b). Low-power view of the central part of the lesion – note the granular structures inside the cell (hematoxylin and eosin stain)|
Click here to view
|Figure 3: (a). High-power (20X) view of the lesion – appreciate the epithelium–connective tissue interface; granule-rich cells making the lesional area. (b): High-power (40X) view of lesional cells (hematoxylin and eosin stain)|
Click here to view
| Discussion|| |
GCTs have been reported from several sites including the skin, subcutaneous tissue, larynx, trachea, bladder, uterus, vulva, and central nervous system. However, they are most common in the tongue with literature reports of about 30% to 60% of all the GCTs. As per the last large-scale systematic review in 2018, there were 1499 cases from 47 studies and 10.47% of all cases occurred in the tongue. This report indicated that the patient presented with an asymptomatic nodule and in 12.46%, there was associated history of trauma or chronic inflammation. In the present case too, the GCT presented as an asymptomatic, slow-growing nodule and there was a history of minor trauma because of orthodontic appliances, as claimed by the patient. In the literature, there is one isolated case wherein the GCT arose with the placement of orthodontic appliance. However, the site in that particular case was a lateral border of the tongue, a site more prone for repeated iatrogenic injury emanating from the orthodontic appliance. In the present case report, the site is the dorsal surface, a site that is relatively immune to injuries from orthodontic appliance.
Clinically, most of the GCTs are slow-growing, solitary, firm, whitish, painless, submucosal nodules with a smooth or ulcerated surface. In the present case too, the lesion was in concurrence with such a report. The usual age of reporting was the fourth to sixth decade, whereas the present case was a young female. Histopathologically, the GCTs are not encapsulated and extend into the associated skeletal muscle. In the present case too, we did observe this phenomenon.,, Traditionally, GCTs are reported to take intense eosinophilic stain and often associated with pseudoepitheliomatous hyperplasia of the overlying epithelium in up to 87% of cases, whereas the present case did not have such a histological picture. This pseudoepitheliomatous hyperplasia, owing often to epithelial reaction, often masquerades as glossal squamous cell carcinoma leading to mis-diagnosis. In the present case, there was no such histological picture, and additionally, excisional biopsy was sufficiently deep to identify the pathognomonic granular cells avoiding the trap of potential mis-diagnosis.,, The clinical presentation could mislead the diagnosis, and there have been instances where there was a squamous cell carcinoma identified to be associated with a GCT. Hence, a careful approach is warranted.,,
The treatment for small GCTs is conservative surgical excision. A complete resection, given the absence of capsules, would favor a low recurrence rate as incomplete removal is not feasible because of poorly defined margins of a GCT., An astute surgeon would want to ensure that the excision or resection margins are free of lesion, which is not feasible with GCTs. Hence, a conservative removal with frequent follow-up is essential to assess the complete prognosis. Recently, LASER ablation of lesion has been reported. Till the efficacy of the LASER is established, the gold standard treatment would be surgery.
Orthodontic treatment by itself may predispose to certain oral lesions such as white spot lesions and acute and chronic traumatic or reactive lesions. Some of these lesions could be precipitated by trauma such as orthodontic appliance. The literature has one GCT report that was associated with active orthodontic appliance, though it was in the lateral border of the tongue. In the present case, the lesion was on the dorsal surface of the tongue, an area immune to the damage caused by the orthodontic appliance; hence, the etiology in the present case cannot be fully attributed to the trauma alone.
| Conclusion|| |
A rare case of GCT in a young female is reported along with its management. The exact role of trauma and inflammation in etiopathogenesis of GCT warrants further investigation.
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
Conflicts of interest
There are no conflicts of interest.
| References|| |
Mobarki M, Dumollard JM, Dal Col P, Camy F, Peoc'h M, Karpathiou G. Granular cell tumor a study of 42 cases and systemic review of the literature. Pathol Res Pract 2020;216:152865. doi: 10.1016/j.prp. 2020.152865.
Ferraz PD. Granular cell tumor (Abrikossoff's tumor) of the tongue: A case report. J Cancer 2021;6:9.
Hita-Davis P, Edwards P, Conley S, Dyer TJ. Granular cell tumour of the tongue in a 17-year-old orthodontic patient: A case report. Oral Surg 2014;7:93-9.
Nacouzi M. Granular cell tumor of the tongue: A rare case presentation of a 14 years old girl. Jpn J Clin Med Res 2021;1. doi: 10.47363/JJCMR/2021 (1) 111.
Bedir R, Yilmaz R, Ibrahim Sehitoglu AO. Coexistence of granular cell tumor with squamous cell carcinoma on the tongue: A case report. Iran J Otorhinolaryngol 2015;27:69-74.
Ferreira JC, Oton-Leite AF, Guidi R, Mendonça EF. Granular cell tumor mimicking a squamous cell carcinoma of the tongue: A case report. BMC Res Notes 2017;10:1-6.
Krishnamurthy A, George R, Majhi U. Malignant granular cell tumor of the tongue: A clinico-pathological challenge. Indian J Surg Oncol 2014;5:71-4.
Viani MV, Corcione L, Di Blasio C, Bologna-Molina R, Vescovi P, Meleti M. A single case report of granular cell tumor of the tongue successfully treated through 445 nm diode laser. In Healthcare 2020;8:267. doi: 10.3390/healthcare 8030267.
Dr. S M Balaji
Director and Consultant Oral and Maxillofacial Surgeon, Balaji Dental and Craniofacial Hospital, 30, KB Dasan Road, Teynampet, Chennai - 600 018, Tamil Nadu
Source of Support: None, Conflict of Interest: None
[Figure 1], [Figure 2], [Figure 3]