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    Table of Contents - Current issue
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March 2021
Volume 25 | Issue 4
Page Nos. 1-112

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CASE REPORTS  

Nasopharyngeal melanoma: An unusual entity Highly accessed article p. 1
Sobhna Mattoo, Sheenam Azad, Brijesh Thakur, Seema Acharya
DOI:10.4103/jomfp.JOMFP_63_20  
Nasopharyngeal melanoma is a rare condition with an estimated incidence of about 0.3/10 lac per year and has a slight female predominance. It can arise from stem melanocytes and mature melanocytes of the submucosa that have acquired genetic alterations, may be due to tobacco, trauma and oxidative stress. It resembles other common polypoidal lesions; therefore, histopathological examination with immunohistochemistry plays a pivotal role in confirming the diagnosis. Lack of specific clinical features often leads to a delay in diagnosis.
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Parapharyngeal space pleomorphic adenoma with extensive lipometaplasia – A rare unique pathological entity p. 4
Suresh Mani, Ronald Anto, Rajiv C Michael, Madhurima Ponmar
DOI:10.4103/jomfp.JOMFP_117_20  
Pleomorphic adenoma (PA) is the most common neoplasm of the salivary gland. Fat-containing tumors encountered in salivary glands are very rare. We had a case of PA of parapharyngeal space and surprisingly the final histopathology showed an unusual finding of lipometapasia. We believe that this is the first case from parapharyngeal space to be reported in the literature. The aim of this report is to discuss the behavior of lipometaplasia in PA with review of literature. Pathological characterization of this rare tumor might help in proper classification and enhance their recognition.
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Vanishing bone disease: An enigma Highly accessed article p. 7
Fatema Yusuf Saify, Suchitra Gosavi, Shilpa Jain, Meenakshi Sood
DOI:10.4103/jomfp.JOMFP_112_20  
Vanishing bone disease is a rare clinical entity with unknown etiology. This disease affects individual irrespective of age or sex. Various names have been used in the literature to describe this condition such as Gorham's disease, phantom bone disease, massive osteolysis, disappearing bone disease and acute spontaneous absorption of bone. The pathogenesis is unknown and the treatment still remains controversial. Considering the rarity of the disease, we report here an interesting and unique case of massive osteolysis of the lower jaw that affected the mandibular basal and alveolar bone. The diagnoses lead on the association of clinical, radiological and histological features.
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Orthokeratinized odontogenic cyst: Report of eight cases and review of literature regarding its malignant transformation p. 11
Nazanin Mahdavi, Mona Zavarei, Samira Derakhshan, Mahboube Hashemi Nasab
DOI:10.4103/jomfp.JOMFP_1_20  
Orthokeratinized odontogenic cyst (OOC) is an uncommon odontogenic cyst. It has been categorized as a subtype of odontogenic keratocyst (OKC). In 2005, it was classified as a distinct entity. OOC should be histopathologically differentiated from OKC, which has a higher recurrence rate and lower malignant potential. In addition, OOC should be examined for malignant transformation. The epithelium of odontogenic cysts may rarely show malignant transformation. However, malignant transformation has been reported in inflammatory cysts such as the residual cyst and periapical cyst. The number of carcinomas arising from an OOC is low. This paper describes eight cases of OOC; out of which, two showed the development of squamous cell carcinoma from their epithelial lining.
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Solitary extramedullary plasmacytoma of the parotid gland in a patient with mixed connective tissue disease p. 18
Dimitris Tatsis, Damianos Sotiropoulos, Aggeliki Baliaka, Konstantinos Vahtsevanos
DOI:10.4103/jomfp.JOMFP_319_20  
Solitary extramedullary plasmacytomas of the head and neck region are rare entities. Very few have been described in the parotid gland. They can clinically and radiologically mimic the rather common benign tumors of the parotid gland (pleomorphic adenoma and Wharthin's tumor), and subsequently the need careful consideration by the involving surgeon and pathologist is needed for a proper diagnosis. In the present study, a case of solitary extramedullary plasmacytoma in the left parotid gland of a 55-year-old patient with mixed connective tissue disease is reported, along with the relevant clinical, imaging, operative, and histopathological findings. Postoperative hematological investigation to confirm the singularity of the lesion was performed. Complementary treatment with radiotherapy was followed. Disease-free, 1 year follow up is also presented.
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Clear-cell variant of oral squamous cell carcinoma: A rare entity p. 22
Pratibha Ramani, S Gheena, Monika Karunagaran, R Hannah
DOI:10.4103/jomfp.JOMFP_295_20  
Clear-cell variant of oral squamous cell carcinoma is an extremely rare entity in the maxillofacial region. We report a case of 42-year-old female who presented with a soft-tissue growth with erythematous and nonscrapable irregular white patches on the left alveolar mucosa for the past 3 months. Histopathologic examination showed lobules and sheets of clear cells with features of malignancy extending from the surface epithelium. Periodic acid–Schiff and mucicarmine stains showed a negative reaction. Immunohistochemical study using antibody for pan-cytokeratin revealed intense positivity and negative for the markers such as S-100, smooth muscle actin and CD 117.
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Langerhans cell histiocytosis: A diagnostic enigma in the oral cavity p. 27
Latha Mary Cherian, Dhanya Sasikumar, Pradeesh Sathyan, Binuja Elsa Varghese
DOI:10.4103/jomfp.JOMFP_296_20  
Langerhans cell histiocytosis (LCH) is a rare reactive and proliferative disease of histiocytes. The disease occurs predominantly in children and rarely in adults. This disease of unknown etiology exhibits extreme clinical heterogeneity. Even though LCH manifests initially in the oral cavity in most of the cases, owing to the relative rarity of the condition, it remains a disease in which the diagnosis is often delayed, missed or misdiagnosed. This is a case of LCH in a child which presented with swelling in the mandibular region.
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Hybrid odontogenic tumor masquerading as a salivary gland lesion: A diagnostic predicament p. 32
Sudip Indu, Anubhuti Sood, Deepika Mishra
DOI:10.4103/jomfp.JOMFP_51_20  
Hybrid odontogenic tumors are sporadic, where the distinctive areas of more than one odontogenic tumor tissue type have been reported. The occurrence of adenomatoid odontogenic tumor (AOT) with calcifying epithelial odontogenic tumor (CEOT) like areas histologically simulating salivary gland pathology is an unusual finding that has not been previously reported in the literature. We report the case of a 32-year-old female presenting with slow-growing firm swelling, radiographically as a pear-shaped radiolucent lesion in the interdental region of maxillary incisors. Histologically, the tissue showed nests and anastomosing strands of the bland cuboidal to squamoid epithelial cells showing nuclear pleomorphism, hyperchromatism, and abundant cytoplasm with prominent intercellular bridges focally. Multiple basophilic calcifications, amyloid-like material, duct-like formation, and mucinous spillage are seen. Tumor cells showed immunopositivity for CK 7, CK 19, CK 8/18 and low Ki67, p63, and immunonegativity for S100 suggesting of a hybrid lesion of CEOT with AOT.
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Mucocele of the tongue: A case report and review of literature Highly accessed article p. 37
Saurabh R Nagar, Gabriela Fernandes, Anuradha Sinha, Kamil N Rajpari
DOI:10.4103/jomfp.jomfp_396_20  
Mucoceles are common cystic lesions in the oral cavity. It may occur in different locations in the oral mucosa due to trauma or obstruction of minor salivary gland ducts with the lower lip as the predominant site. However, mucoceles located on the ventral surface of the tongue originating from the anterior lingual salivary glands are extremely rare and often overlooked during screening procedures because of their asymptomatic nature. Here, we report an interesting case of mucocele on the anterior ventral surface of the tongue in an 11-year-old female based on the clinical and histopathological diagnosis. Moreover, mucoceles should be considered as one of the differential diagnoses while evaluating a growth involving the ventral surface of the tongue in young female children.
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Traumatic ulcerative granuloma with stromal eosinophilia: A puzzle p. 42
Bhagirathi D Lakkam, Madhusudan Astekar, Shakir Alam, Adeeba Saleem
DOI:10.4103/jomfp.JOMFP_321_20  
Traumatic ulcerative granuloma with stromal eosinophilia (TUGSE) is an ulcerative condition of the oral mucosa amid a chronic course and delayed healing. It is a reactive lesion that usually affects the tongue. TUGSE is a rare mucosal lesion to aid as an ulcer or an indurated submucosal mass and might cause diagnostic problems as it mimics a traumatic or neoplastic ulcer. However, chronic irritation from traumatic agents is well thought out to be a major initiating aspect. Histopathologically, it is categorized by an eosinophilic inflammatory infiltrate penetrating into the underlying muscle. It is significant to make out this mucosal lesion as it mimics malignant ulcers; the condition tends to resolve on its own. Thus, attentiveness of this entity is imperative to emphasize the accurate diagnosis of indurated ulcerated lesions and convey suitable and effective treatment. The present case highlights the clinical aspects, etiopathogenesis and histopathology of this unusual lesion.
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Epstein–Barr virus-positive ulcer in the oral cavity p. 46
Jose David Ramos-Baena, Luis Fernando Jacinto-Alemán, Elba Rosa Leyva-Huerta, Javier Portilla-Robertson
DOI:10.4103/jomfp.JOMFP_26_19  
Epstein–Barr virus-positive ulcer (EBV + U) is a recently reported B cell lymphoproliferative disorder in the oral cavity, oropharynx, gastrointestinal tract and skin, principally in immunosuppressed patients. A 53-year-old female patient with rheumatoid arthritis treated with methotrexate, presenting ulcers of unknown duration on the dorsum and the lateral left border of the tongue. Excisional biopsy, histopathological analysis and histochemical stains for syphilis (Warthin–Starry), mycotic diseases (Grocott silver methenamine), tuberculosis (Ziehl–Neelsen), immunohistochemistry tests for herpesvirus type 8 (CMV), EBV (LMP-1) and DNA extraction for polymerase chain reaction (PCR) assay to CMV, EBV and herpes simplex virus-1 were performed. Posterior to PCR assay, the final diagnosis was EBV + U in the oral cavity. Acyclovir® was prescribed, showing clinical improvement. A case of EBV + U with clinical characteristics similar to other lesions or conditions has been reported. Special assays are necessary for an accurate diagnosis and treatment.
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Nonsyndromic delayed eruption of multiple teeth: A rare case report p. 51
Valen Dela D'souza, Prasanna Kumar Rao, Raghavendra Kini
DOI:10.4103/jomfp.JOMFP_323_20  
Dental eruption is a very finely regulated process. A delay in tooth eruption may be due to a disturbance caused by local, systemic, or genetic abnormalities. Delayed eruption of multiple teeth in the absence of any etiology is very rare. Here, we report a case of delayed eruption in a 16-year-old female patient with multiple congenitally missing teeth and bilaterally ankylosed deciduous teeth with no underlying systemic or genetic disease.
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An atypical presentation of plasma cell gingivitis with generalized skin lesions p. 54
Gaurav Nitin Ketkar, G Kaarthikeyan
DOI:10.4103/jomfp.JOMFP_292_20  
This article aims to present a report of an atypical clinical presentation of a plasma cell gingivitis (PCG) case with unusual skin lesions. PCG is a rare benign inflammatory condition which can be classified into Type 4 hypersensitivity reaction. It occurs due to reaction to unknown antigen, often flavoring agents or spices found in chewing gums, toothpastes and lozenges. Histologically, the lesion shows dense plasma cells infiltrate in the connective tissue. Early diagnosis of PCG is essential to differentiate from variety of conditions, namely, leukemia, HIV infection, discoid lupus erythematosus, atrophic lichen planus, desquamative gingivitis, or cicatricial pemphigoid which must be differentiated through hematologic and serologic testing. In this article, we will discuss a case of PCG with unusual skin lesions.
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Nasopharyngeal carcinoma ex-pleomorphic adenoma (noninvasive): A report of a rare case p. 58
Sujata Naik, Prateek Das, Rashmi Patnayak, Santosh Kumar Swain
DOI:10.4103/jomfp.JOMFP_241_20  
Pleomorphic adenoma (PA) is the most common type of benign tumors of minor salivary glands. A carcinoma ex-pleomorphic adenoma (CXPA) is a malignant epithelial neoplasm originating from either a primary or recurrent benign PA. The nasopharynx is an extremely uncommon location for this tumor. A 32-year-old male had complaints of nasal blockage. In noncontrast computed tomography, a soft-tissue mass was present in the nasopharynx. Histopathological and immunohistochemical examination of the endoscopically excised mass revealed features of CXPA, noninvasive in nature. Careful histopathological examination is the key to identify this uncommon entity. To the best of our knowledge, <20 cases have been published so far.
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“Migratory Kerato osteomyelitis” – A refractory lesion of the mandible: A pathologist's and surgeon's dilemma!! p. 61
Sudip Indu, Narendra Babu
DOI:10.4103/jomfp.JOMFP_138_20  
A 62 year old female patient presented with a chief complaint of non healing extraction socket in the lower left back teeth region. She underwent extraction of 37 at a private clinic following which an ulceroinfiltrative lesion developed at operated side. Multiple biopsies from the lesion were inconclusive. Computed tomography revealed an osteolytic lesion radiologically simulating carcinoma or a chondrosarcoma. Because of persistent debilitating symptoms the patient underwent Wide local excision (WLE) with left segmental mandibulectomy. Frozen sections were negative for malignancy. All margins were free from malignancy. Lymph nodes dissected showed reactive morphology. Ziehl–Neelsen, Periodic acid-Schiff and Gram stain were all negative. The lesion continued to spread even after successful surgical intervention with adequate surgical margin. The patient was eventually lost because of severe cardiac arrest during her last surgical intervention for mandibular arch reconstruction. As the lesion migrated from one side of the mandible to the other, kept showing clinical features of inflammation of the bone and bone marrow simulating features of osteomyelitis and the continued presence of keratin histologically, we contemplated whether we could give a nomenclature to the lesion as that of “Migratory Kerato osteomyelitis of the oral cavity.” This could be one of case of a lifetime where multiple consultations with oral and general pathologists failed to reach a conclusive diagnosis!
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Concurrent dentigerous cyst with ossifying fibroma of the mandible p. 68
Bhavana Gupta, Narendra Nath Singh, Ankita Tandon
DOI:10.4103/jomfp.JOMFP_85_20  
Dentigerous cyst (DC) and ossifying fibroma (OF) are intraosseous lesions of the jaw. Both are varied pathological entities with a wide spectrum of clinical and histological features along with distinct treatment plan and prognosis. While OF comes under fibro-osseous lesions of the jaws, DC is a developmental odontogenic cyst which is formed by the accumulation of fluid between reduced enamel epithelium and enamel or between layers of the enamel organ. This case report presents a rare display of two distinct pathologies synchronously and aims to discuss the possible histogenesis for the same.
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Bilateral gemination of permanent maxillary canine with labial and palatal talon's cusps: A rare entity p. 71
DB Nandini, BS Deepak, Deepak Ningombam Singh, P Aparnadevi
DOI:10.4103/jomfp.JOMFP_118_20  
Gemination is a morphologic anomaly of the tooth characterized by the formation of a macrodont. Bilateral presentation is rarely reported in permanent canines. Talon's cusp is an accessory cusp projecting lingually from the cingulum to the incisal edge and may be seen facially sometimes. Simultaneous occurrence of facial and lingual talons on the same tooth is rare. Concurrent occurrence of all the three entities has not been reported. These anomalies can cause unpleasant esthetic appearance due to irregular morphology. The presence of deep grooves on these teeth increases the susceptible to caries and periodontal disease requiring an endodontic intervention. Proper clinical and radiographic examination provides an accurate diagnosis and helps the clinician in the proper treatment planning and avoiding further complications. This article presents a unique case of simultaneous occurrence of bilateral gemination in the permanent maxillary canines with labial and palatal talon's cusps, which has not been reported in the literature till date.
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Dentinogenesis imperfecta type II: Diagnosis, functional and esthetic rehabilitation in mixed dentition p. 76
Ramneet Kaur, Asha Karadwal, Deepak Sharma, Manpreet Kaur Sandhu
DOI:10.4103/jomfp.JOMFP_172_20  
Dentinogenesis imperfecta (DGI) type II affects both primary and permanent dentitions and has the autosomal mode of inheritance. The affected teeth may appear as amber or gray because of chipping of enamel shortly after their eruption. Correct diagnosis and management are highly needed to restore the quality of oral health and to improve esthetics and masticatory functions. We present here a case of systematic and conservative dental approach in the management of a 7-year-old child having Dentinogenesis Imperfecta Type II (DGI Type II) with 1 ½ follow-up.
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Primary lymphangioma of palatine tonsil p. 81
Ranjana Giri, Goutami Dasnayak, Sarojini Raman, Urmila Senapati
DOI:10.4103/jomfp.JOMFP_203_20  
Lymphangiomas are benign congenital tumors of the lymphatic system. Tonsillar lymphangiomas are extremely rare. Histopathological confirmation is essential to make the correct diagnosis. We report a case of primary lymphangioma of the left tonsil in a 17-year-old male patient who presented with difficulty in swallowing and was clinically diagnosed as a tonsillar cyst.
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Developing complex odontoma in a 4-year-old child with active ameloblastic follicles: A case report p. 84
Harikrishnan Prasad, Gopal Shiva Kumar, Muthusamy Rajmohan, Korath Varkey
DOI:10.4103/jomfp.JOMFP_249_20  
The World Health Organization (WHO) classification of odontogenic tumors, in its different iterations, has seen several modifications in benign mixed odontogenic tumors. Odontoma is a benign mixed odontogenic tumor that shows dental hard tissue formation to varying degrees. Although odontomas are the most common odontogenic tumors, they are mostly seen in the form of compound odontomas, where the dental hard tissues resemble tooth-like structures. Complex odontoma shows a haphazard mass of dental hard tissues and is generally identified at an older age (mean age of around 20 years). Incidence of complex odontoma in very young children is quite rare, with only around 10 cases associated with the primary dentition being reported in the literature till date. Unlike other odontomas, our case showed an active epithelial component in the form of tooth buds and ameloblastic follicles along with the hard tissues, which made it difficult to distinguish from other similar lesions like ameloblastic fibroma with hard tissue formation and ameloblastoma arising from an odontoma. This article reports the 11th case of complex odontoma associated with the primary dentition, and the youngest such case to be reported in the Indian population.
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Gingival salivary gland choristoma: An unusual case report p. 90
Mario Héctor Torres Medina, RamÓn Franco Topete, Mario Nava Villalba, José Sergio Zepeda Nuño
DOI:10.4103/jomfp.JOMFP_284_19  
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.
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An unusual presentation of an odontogenic tumor: A diagnostic quandary p. 94
Priya Thomas, Aneesh Sebastian, Rekha Krishnapillai, Sapna Chandran
DOI:10.4103/jomfp.JOMFP_147_20  
Squamous odontogenic tumor (SOT) is a rare benign neoplasm and may be located to multiple sites in the oral cavity mouth. As per the literature, there have been <50 reported cases. The tumor is often asymptomatic, although it can present with symptoms of pain and tooth mobility. Peripheral odontogenic tumor is a rare entity derived from either epithelial or mesenchymal portions of the tooth-forming apparatus. Lesions are common to gingiva and alveolar mucosa. Peripheral granular cell ameloblastoma (GCA) is considered to be even rarer. The purpose of the study is to report a case of SOT with a synchronous association with peripheral GCA of cystic nature in the mandible. The occurrence of SOT with ameloblastoma has not been reported as per the literature search.
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Ghost cell odontogenic carcinoma of anterior mandible: A rare case report with review of literature p. 99
Gopikrishnan Vijayakumar, Mala Kamboj, Anjali Narwal, Anju Devi
DOI:10.4103/jomfp.JOMFP_195_20  
A 24-year-old male reported to the outpatient department with a complaint of swelling of the anterior lower jaw region for 9 months with history of traumatic injury and extraction of teeth from the same region, a month before the onset of swelling. Swelling was obvious extra- and intraorally which on examination presented as a soft to firm non-tender and non-fluctuant mass with an approximate size of 4 cm × 3 cm, extending from 34 to 43 region with obliteration of labial vestibule. Panoramic radiograph and cone-beam computed tomography showed a well-defined radiolucency in the mandibular anterior region crossing the midline with erosion of labial bony plates and root of 42 along with a tooth-like radiopaque mass within the lesion. Provisional diagnoses of odontogenic keratocyst, ameloblastomas, central giant cell granuloma and calcifying epithelial odontogenic tumor were listed. The histopathological and immunohistochemical examination of lesion followed by the biopsy confirmed the diagnosis of Ghost cell odontogenic carcinoma.
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Pediatric leiomyoma of the oral cavity: A rare entity p. 109
Srujan Kumar Peruka, Uday Kiran Uppada, Gireesha Reddy Dawalgari
DOI:10.4103/jomfp.jomfp_412_20  
Leiomyoma is a benign smooth muscle tumor that frequently occurs in the uterine myometrium of the uterus and gastrointestinal tract rarely but seldom encountered in the oral cavity, particularly in children. Its occurrence in the oral cavity is considered rare probably because of the scarcity of smooth muscle tissue in the oral cavity. The frequently affected anatomical areas intraorally are lips, palate and tongue. The purpose of this case report is to put forth a rare case of oral leiomyoma in a 10-year-old girl child involving the retromolar region.
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Journal of Oral and Maxillofacial Pathology | Published by Wolters Kluwer - Medknow
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