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 Table of Contents  
CASE REPORT
Year : 2016  |  Volume : 28  |  Issue : 1  |  Page : 39-43

An unveiled innocuous oral ulcer: Report of a rare case


1 Department of Oral Medicine and Radiology, Maulana Azad Institute of Dental Sciences, New Delhi, India
2 Department of Oral Medicine and Radiology, ESIC Dental College, New Delhi, India
3 Department of Shalakya (Ayurveda Eye and ENT), Tibbia College and Hospital, New Delhi, India

Date of Web Publication8-Sep-2016

Correspondence Address:
Khushboo Singh
Department of Oral Medicine and Radiology, Maulana Azad Institute of Dental Sciences, New Delhi - 110 002
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0972-1363.189986

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   Abstract 

Mucoepidermoid carcinoma (MEC) is the most common primary salivary gland neoplasm frequently affecting the parotid gland. We report a case of MEC in a 24-year-old male in the retromolar area, which on initial examination gave impression of an innocuous-appearing traumatic ulcer but on histopathological examination turned out be an aggressive MEC. This article also shows the role of traditional therapy with Ayurveda in the management of this case. However, there are no previous scientific evidences in literature for treatment with such type of therapy.

Keywords: Alternate medicine, carcinoma, mucoepidermoid, retromolar area


How to cite this article:
Gupta S, Jetly V, Singh K, Chhoker RK. An unveiled innocuous oral ulcer: Report of a rare case. J Indian Acad Oral Med Radiol 2016;28:39-43

How to cite this URL:
Gupta S, Jetly V, Singh K, Chhoker RK. An unveiled innocuous oral ulcer: Report of a rare case. J Indian Acad Oral Med Radiol [serial online] 2016 [cited 2022 Jul 3];28:39-43. Available from: https://www.jiaomr.in/text.asp?2016/28/1/39/189986


   Introduction Top


Salivary gland carcinomas (SGC) are rare neoplasms that account for less than 1% of all human cancers.[1],[2] Among SGCs, the mucoepidermoid carcinoma (MEC) is the most common primary neoplasm, representing approximately 30% of salivary malignancies.[2],[3] The tumor occurs most frequently in the parotid gland, followed by minor and submandibular salivary glands.[4] Intraorally, palate (45%) is the preferred soft tissue site, however, the involvement of the retromolar/tonsillar region is quite common.[5] Intraosseous tumors have been reported in both maxilla and mandible comprising 2–3% of all MECs.[6] As the name implies, the tumor is composed of both mucus-secreting cells and epidermoid-type cells in varying proportions and is characterized histopathologically into high, low, or intermediate grade. We report a case of MEC in a 24-year-old male in the retromolar area, which on initial examination gave the impression of an innocuous appearing traumatic ulcer, but on histopathological examination turned out be an aggressive MEC.


   Case Report Top


A 24-year-old male patient presented to our department in June 2008 with a complaint of mobile anterior teeth. The patient had undergone trauma to the upper anterior region 15 days back which led to avulsion of his one upper anterior tooth and mobility of other anterior teeth. Extraoral examination revealed no abnormality and no significant lymphadenopathy. Intraoral examination revealed avulsion of 11 and grade I mobility of 12, 21, and 22. Vitality test was done in upper anteriors which were vital. In addition, the patient had buccally impacted 28 with a small area of ulceration surrounded by keratosis in relation to the left retromolar area, of which he was not aware. Thus, a provisional diagnosis of traumatic ulcer due to impacted 28 was made. The patient was advised extraction of 28 with a topical application of 2% chlorhexidine gluconate for the traumatic ulcer and was kept on follow-up. Patient was reviewed 2 weeks after extraction of 28 and it was observed that there was no improvement in appearance of ulcer. Thus, an incisional biopsy was carried out from the retromolar area and sent for histopathological examination.

The biopsy specimen showed moderate dysplasia. The underlying stromal tissue showed infiltration by tumor cells arranged in nests and islands. Few vacuolated cells were noted along with epithelial cells with hyperchromatic enlarged nuclei [Figure 1] and [Figure 2]. A special stain with mucicarmine was done which was positive in scattered vacuolated cells [Figure 3]. Overall features were suggestive of MEC. Contrast enhanced computed tomography (CECT) was performed to determine the extent of the lesion. It revealed a poorly circumscribed heterogeneously enhancing mass lesion in the left retromolar trigone causing destruction of adjacent maxillary alveolar arch and invasion into left medial pterygoid and left temporalis with no lymphadenopathy [Figure 4]. Patient was referred to the Ear, Nose, and Throat department for further evaluation and management. The treatment involved excision of the primary tumor with a 2 cm margin with left segmental mandibulectomy with supraomohyoid neck dissection (SOND). The intraoral defect was closed using fat in the retromolar region. The excisional biopsy suggested a high grade MEC reaching close to inferior, superior, and anterior resected margins. The lateral and posterior margins were free of tumor. Along with the tumor, a fibromuscular tissue of 6 × 3 × 1.5 cm in dimension was excised which showed salivary gland and skeletal muscle free of tumor. Out of the seven lymph nodes isolated, one showed tumor metastasis. The patient was referred to the Oncology department for the management of the residual tumor. A whole body positron emission tomography (PET) scan was carried out which showed evidence of focal fluoro-deoxyglucose (FDG) avidity along the anterior resected margin of the mandible. Mildly FDG avid 1.2 × 1 cm sized right level II lymph node and tiny non-FDG avid right supraclavicular lymph nodes were also noted. PET findings suggested residual/recurrent tumor in the anterior resected margin of the mandible [Figure 5] and [Figure 6]. Hence, it was decided to administer chemotherapy followed by radiotherapy. After the first cycle of chemotherapy, the patient was not able to tolerate the side effects and discontinued the treatment. Through some contacts, the patient came to know of a traditional Ayurvedic practitioner who is known to treat many difficult cases of malignant lesions. Patient initiated the Ayurvedic treatment and started feeling better with that.
Figure 1: Histopathology (Low power): Large number of epithelial cells alongwith vacuolated cells arranged in whorls

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Figure 2: Histopathology (High power): Predominantly hyperchromatic epithelial cells

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Figure 3: Histopathology: Mucicaramine positive scattered vacuolated cells

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Figure 4: Computed tomography scan: Poorly circumscribed heterogeneously enhancing mass lesion in the left retromolar trigone causing destruction of the adjacent maxillary alveolar arch and invasion into the left medial pterygoid and left temporalis muscle

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Figure 5: Positron emission tomography-computed tomography: Excised tumor mass along with the resected mandible on the left side

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Figure 6: Fluoro-deoxyglucose positron emission tomography scan: Focal flurodeoxyglucose activity along the anterior resected margin of the mandible

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The details of the Ayurvedic treatment regarding the drug therapy received are mentioned below:

  1. Local therapy: Chewing a betel leaf containing leaf bud of Arka (Calotropis procera) for half an hour with empty stomach in morning, swallowing the juice, and spitting out the residual part
  2. Wheat Grass juice (Triticum aestivum L.): Drinking the juice empty stomach early morning which was made up of 100 g of wheat grass leaves in 200 ml of water with a pinch each of roasted powder of black cumin and black salt
  3. Decoction prepared from the following herbs:




  1. Ghritkumari (Aloevera): 1.5 kg, fresh
  2. Indravaruni fruit pulp (Citrullus colocynthis): ½ kg, dry
  3. Giloy (Tinospora cordifolia): ½ kg, fresh
  4. Makoya (Solanum Nigrum): ½ kg, dry (whole plant).


Dosage of decoction: 25 ml of decoction was taken after each meal, i.e., twice to thrice per day.

The abovementioned three therapies were continued for approximately 4.5 months.

4. Miscellaneous therapy: After 1 month of treatment, patient complained of facing difficulty in opening the mouth. So, patient was advised the following treatment in the form of powdered herbs in the manner and dosage mentioned as follows:



  1. Akarkara (Analycus pyrethrum): 25 g
  2. Salam Panja (Eulophia campestris): 25 g
  3. White Musli: 25 g
  4. Saunf: 25 g
  5. Sugar: 25 g.


All of the abovementioned components were mixed and powdered fine.

Dosage: 1 teaspoon of this mixture was taken with milk at bed time.

After a 4 month course of the Ayurvedic treatment, a repeat PET scan was done to determine the status of the residual tumor. In comparison to the previous PET scan, there was complete resolution of the hypermetabolism along the anterior resected margin of the left side of the mandible [Figure 7]. The patient is kept on close follow-up since the last six years and is doing fine [Figure 8].
Figure 7: Fluoro-deoxyglucose positron emission tomography scan: Complete resolution of the hypermetabolism along the anterior resected margin of the left side of the mandible

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Figure 8: Intraoral views of 8 month post-treatment follow up

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   Discussion Top


MEC is a malignant epithelial tumor which accounts for 6–8% of all salivary gland tumors. It is generally believed that the tumor arises from reserve cells of the excretory ducts of the major/minor salivary glands, whereas intrabony lesions are believed to arise from embryologic entrapped salivary gland tissue or transformation of odontogenic cystic lining.[5] The tumor is classified clinically as high or low grade depending on the clinical aggressiveness. The tumor of low grade malignancy usually appears as a slowly enlarging, painless mass which stimulates pleomorphic adenoma.

The tumor of high grade malignancy grows rapidly and produces pain as an early symptom. Facial nerve paralysis is frequent in parotid tumors. In intraoral tumors, numbness of the adjacent structures and ulceration may be noted. Distant metastasis to the lung, bone, brain, and subcutaneous tissues are also common. Histologically, MEC is composed of mucous-secreting cells, epidermoid-type cells, and intermediate cells. Radical surgery is the treatment of choice. Adequate excision is important in all grades of tumor with high recurrence reported with positive surgical margins, which is 80% for high grade tumors.[1] Few studies have assessed the role of chemotherapy and showed that high grade tumors may show sensitivity similar to SCC.[7]

Our reported case was quite unusual as it was an incidental finding of MEC of retromolar area in a 24-year-old male, which clinically gave the impression of a traumatic ulcer. Therefore, it is important to carry out biopsy procedure in all the cases whenever there is persistent nonhealing ulcer even after the removal of the stimulating factor. Although our case did show the efficacy of ayurvedic treatment, there are no previous scientific data which have proven the efficacy and role of ayurvedic treatment modalities. However, some of the review articles and in vivo and in vitro studies support their role. Anacyclus pyrethrum (Akarkara) has immunostimulant activity on cyclophosphamide-induced immunosuppression in rats.[8] Thymoquinone extracted from Nigella sativa (black cumin) has antioxidant role and improves the body's defense system, induces apoptosis, and controls Akt pathway.[9]Triticum aestivum Linn (wheat grass) has been shown to possess anticancer activity, antiulcer activity, antioxidant activity, antiarthritic activity, and blood building activity. It has antioxidant potential which is derived from its high content of bioflavonoids such as apigenin, quercitin, and luteoline, and it has also the property of inhibiting the proliferation of leukemic cells.[10]

Ethanolic and sapogenin extract of Chlorophytum borivilianum (white musli) roots possess immunomodulatory, antitumor, antimutagenic, and chemomodulatory activity.[11]Giloy satva (Tinospora cordifolia Linn known as Guduchi) and hydroalcoholic extract of the plant Curculigo orchioides Linn (kali musli) have antioxidant properties. Dried latex of Calotropis procera has the potential for anticancer therapy; Calotropis procera protein (CP-P) inhibits the proliferation and induced apoptosis of breast cancer cells through the suppression of nuclear factor kappa B (NF-κB) activation.[12]Solanum nigrum interferes with the structure and function of tumor cell membrane, disturbs the synthesis of DNA and RNA, changes the cell cycle distribution, and has anticancer abilities by blocking the antiapoptotic pathway of NF-κB.[13]

As we know the effect of chemotherapeutic drugs on MEC is not well-established, the question arises whether the resolution of residual tumor is due to the effect of single cycle of chemotherapeutic drugs or is it the outcome of traditional medicines? The answer is still unclear. This calls for more evidence-based scientific studies to substantiate the results of such therapies and a long-term follow-up of those patients who have taken such treatment for possible recurrences. Our purpose of documenting such therapy in this case report is that it can be proved helpful in future research in terms of some traditional therapies, which have taken a far back seat in the modern era of medicine.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
   References Top

1.
Triantafillidou K, Dimitrakopoulos J, Iordanidis F, Koufogiannis D. Mucoepidermoid carcinoma of minor salivary glands: A clinical study of 16 cases and review of literature. Oral Dis 2006;12:364-70.  Back to cited text no. 1
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2.
Grisanti S. Cetuximab in the treatment of metastatic mucoepidermoid carcinoma of the salivary glands: A case report and review of literature. J Med Case Report 2008;2:230.  Back to cited text no. 2
    
3.
Pires FR, Almeida OP, Araújo VC, Kowalski LP. Prognostic factors in head and neck Mucoepidermoid carcinoma. Arch Otolaryngol Head Neck Surg 2004;130:174-80.  Back to cited text no. 3
    
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Perez DE, Pires FR, Alves Fde A, Lopes MA, Almeida OP, Kowalski LP. Juvenile intraoral mucoepidermoid carcinoma. J Oral Maxillofac Surg 2008;66:308-11.  Back to cited text no. 4
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Kahn MA, Lucas RM. Mucoepidermoid tumor: A case report involving the operculum of an erupting second molar. Oral Surg Oral Med Oral Pathol 1989;68:375-9.  Back to cited text no. 5
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Simon D, Somanathan T, Ramdas K, Pandey M. Central mucoepidermoid carcinoma of mandible: A case report and review of the literature. World J Surg Oncol 2003;1:1.  Back to cited text no. 6
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Rahbar R. Mucoepidermoid carcinoma of the parotid gland in children: A 10 year experience. Arch Otolaryngol Head Neck Surg 2006;132:375-80.  Back to cited text no. 7
    
8.
Sharma V, Thakur M, Singh N, Dixit V. Immunomodulatory activity of petroleum ether extract of Anacyclus pyrethrum. Pharmaceutical Biology 2010;48:1247-54.  Back to cited text no. 8
    
9.
Khan A, Chen HC, Tania M, Zhang DZ. Anticancer activities of Nigella Sativa. Afr J Tradit Complement Altern Med 2011;8:226-32.  Back to cited text no. 9
    
10.
Aydos OS, Ozkan T, Karadag A, Gurleyik E, Altinok B, Sunguroglu A, et al. Antiproliferative, apoptotic and antioxidant activities of wheatgrass (Triticum aestivum L.) extract on CML (K562) cell line. Turk J Med Sci 2011;41:657-63.  Back to cited text no. 10
    
11.
Kumar M, Meena P, Verma S, Kumar M, Kumar A. Anti-tumour, anti mutagenic and chemomodulatory potential of Chlorophytum borivilianum. Asian Pac J Cancer Prev 2010;11:327-34.  Back to cited text no. 11
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12.
Choedon T, Mathan G, Arya S, Vijay L, Kumar V. Anticancer and cytotoxic properties of the latex of Calotropis procera in a transgenic mouse model of hepatocellular carcinoma. World J Gastroenterol 2006;12:2517-22.  Back to cited text no. 12
    
13.
An L, Tang JT, Liu XM, Gao NN. Review about mechanisms of anti-cancer of Solanum nigrum. Zhongguo Zhong Yao Za Zhi 2006;31:1225-6.  Back to cited text no. 13
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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8]



 

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