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    Table of Contents - Current issue
Volume 22 | Issue 4 (Supplement)
Page Nos. 1-140

Online since Friday, February 2, 2018

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From the editor's desk Highly accessed article p. 1
T Smitha
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Odontogenic myxoma: A causality dilemma – Report of a nonpareil case and review of literature Highly accessed article p. 2
Sugunakar Raju Godishala Swamy, Sushma Naag, Sumit Bahl, Esther Priyadarshini
Odontogenic myxoma is a benign locally aggressive neoplasm with a sparse prevalence and incertitude histogenesis. They constitute 3%–6% of odontogenic tumors in gnathic bones. It is ubiquitously seen between vicenarian to early quadragenarian group with female proclivity and fondness to the mandibular jaws. They are silent lesions clinically and show myxoid stroma amidst fibrous background. This report highlights central odontogenic myxoma in a 43-year-old male patient and focuses on concepts, differential diagnosis, molecular concepts and treatment aspect.
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Ameloblastoma arising in the wall of dentigerous cyst: Report of a rare entity p. 7
Sudheer Kanth Kondamari, Sravya Taneeru, Venkateswara Rao Guttikonda, Geetha Kumari Masabattula
Dentigerous cyst (DC) is a developmental odontogenic cyst that encloses the crown of an unerupted tooth by expansion of its follicle with accumulation of fluid between the reduced enamel epithelium and the tooth crown and is attached to the neck of the tooth. The lining of DCs shows a potential for neoplastic transformation to ameloblastoma, squamous cell carcinoma, and mucoepidermoid carcinoma. Here, we report a rare case of an ameloblastoma arising in the wall of a DC.
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Aggressive adenomatoid odontogenic tumor of the mandible: A rare case report and review of the literature p. 11
Sana Shaikh, Shivani Bansal, Rajiv S Desai, Islam Ahmad
Adenomatoid odontogenic tumor (AOT) is a relatively rare and distinct odontogenic tumor that is exclusively odontogenic epithelium in origin. Although considerable numbers of reports are available with regard to the clinical and histological spectrum of AOT, very few have highlighted its rare aggressive nature. This article focuses on an AOT causing jaw swelling in the mandibular anterior region with cortical plate expansion and perforation, root resorption and paresthesia. The present case had remarkably unusual clinical and radiographic features that distinguished it from most conventional AOTs and supported its neoplastic nature.
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A curious case of central odontogenic fibroma: A novel perspective p. 16
Chetana Chandrashekar, Subhalakshmi Sen, Venkadasalapathi Narayanaswamy, Raghu Radhakrishnan
We appraise a case of central odontogenic fibroma (COF) with unusual histologic features of entrapped neural elements and mast cells. The presence of mast cells attributed to the release of stem cell factor, the principal growth and activating factor of mast cells. A putative role for C-kit and mast cells in the pathogenesis of COF is described.
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Orthokeratinized odontogenic cyst with calcification: A rare case report of a distinct entity p. 20
Mamata Kamat, Sampada Kanitkar, Uma Datar, Sanjay Byakodi
Orthokeratinized odontogenic cyst (OOC) is a relatively rare odontogenic cyst, distinct from odontogenic keratocyst (OKC). In the 4th edition of WHO Classification of Head and Neck Tumors (2017), OOC has been included as a separate entity in the category of developmental odontogenic cysts. It presents as a unilocular radiolucent lesion in the posterior mandible and is frequently related to impacted teeth, thus mimicking dentigerous cyst. Due to low local aggressiveness and no association with nevoid basal cell carcinoma syndrome, it does not show tendency to recur. When compared to OKC, OOC exhibits substantial number of differences with respect to clinical, pathological and behavioral features and treatment modalities. Hence, recognizing OOC as unique lesion is mandatory to avoid unnecessary overtreatment. This paper aims to report a rare case of OOC associated with impacted tooth, showing calcifications and emphasizes its differences from OKC. Furthermore, the recent concepts about OKC and OOC are discussed.
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Hemangiomatous ameloblastoma: Case report with a brief review p. 24
Aparna Venigalla, Manasa Bojji, Soujanya Pinisetti, Suresh Babburi
Ameloblastoma is a benign epithelial odontogenic tumor with many histological variants. Hemangiomatous ameloblastoma (HA) is a very rare variant which shows unique histopathological features varying from conventional ameloblastoma. We present a case of a 35-year-old female patient with a swelling over right lower back region of jaw, showing mixed radiolucent-opacity. Incisional biopsy showed microscopic features of desmoplastic ameloblastoma showing extensive desmoplasia and compressed odontogenic epithelial islands. Excisional biopsy revealed ameloblastomatous areas with extensive vascular component microscopically. Based on these findings, a diagnosis of HA was made.
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A mixed odontogenic sarcoma: A challenging histopathologic case and brief review of the literature p. 29
Saede Atarbashi-Moghadam, Ali Lotfi, Sepideh Mokhtari
Ameloblastic fibro-odontosarcoma (AFOS) is an extremely rare malignant mixed odontogenic tumor. The ectomesenchymal part of the neoplasm shows malignancy, whereas the epithelial component is rather benign. In addition, small areas with deposition of enamel matrix and dentine material are seen. The rarity of this neoplasm and microscopic similarities with other malignant and benign tumors can lead to diagnostic problems. Here, we describe the histopathologic features of a new case of AFOS of the mandible in a 34-year-old female patient. It is essential for oral pathologists to be familiar with the microscopic features of this rare neoplasm to have a proper diagnosis. This is also the first reported case of AFOS that closely resembles osteosarcoma in some areas.
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Chondrosarcoma of maxilla p. 35
Asha Karadwal, Shailja Chatterjee
Chondrosarcoma is a rare malignant cartilaginous tumor of the head and neck region. It is a slow-growing tumor and is mostly detected in the anterior maxilla and the base of the skull. We report a case of “Grade II Chondrosarcoma” of the posterior right side of the palate invading maxillary sinus and perforating the floor of the orbit.
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Metastatic renal cell carcinoma presenting as maxillary lesion: Report of two rare cases p. 39
Samira Derakhshan, Sedighe Rahrotaban, Nazanin Mahdavi, Faeze Mirjalili
Oral cavity is not a usual site for metastasis, accounting about 1% of all oral tumors. The majority site for metastasis is the mandible and less common the maxilla followed by intraoral soft tissue. These tumors may be the first clinical manifestation of the primary lesion. One of the most important sites of the primary neoplasm that metastasizes to the jaws is kidney. Therefore, considering to this fact that the metastatic tumors always are not seen in a typical site, it is necessary to rule out the metastatic carcinomas to jaws. We presented two cases of metastatic renal cell carcinoma to maxilla with two different clinical features, one of them with an intraosseous lytic lesion and the other one with a large exophytic mass.
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Melanotic neuroectodermal tumor of infancy: A rare case report p. 44
Abhishek Kumar, V Deepthi, Ruchi Aggarwal, Hema Malini Aiyer
Melanotic neuroectodermal tumor of infancy is a rare benign but locally aggressive neoplasm of neural crest origin with a high recurrence rate. It usually affects infants of <1 year of age. Involvement of maxilla of an infant is the most common presentation although cases outside this setting have been reported. We report the case of a 6-month-old male child presenting with intraoral swelling. Radiologically, a tumor was detected in the right maxilla, and wide local excision was done. Histological examination revealed typical biphasic population of cells with deposits of melanin pigment. Immunohistochemistry study was done confirming the diagnosis.
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Aggressive high-grade Ewing's sarcoma of maxilla: A rare case report p. 48
TL Yogesh, Akshay Shetty, Hema Keswani, Diljith Rishi
Ewing's sarcoma (ES) is an uncommon malignancy of childhood and adults that constitutes 6%–8% of all primary malignant tumors and the third-most common tumor after osteosarcoma and chondrosarcoma, frequently involves the mandible among jaw bones. This article presents a rare case of ES of maxilla in a 22-year-old male patient showing extensive lesion into skull base which was confirmed with computed tomography, dilemmatic histopathologic features in H and E which is not a frequent presentation. Histopathologic features showed monotonous round cells with hypo- and hyper-cellular areas, intralesional hemorrhage and necrosis with lesional cells positive for CD99. Although the prognosis is poor, early diagnosis and long-term follow-up can improve the survival. The diagnosis was confirmed by immunohistochemistry where lesional cells were positive for CD 99 and vimentin. ES of maxilla is a rare and aggressive tumor. Hence, early diagnosis, combined therapy, and long-term follow-up are suggested in such cases.
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Adenoid cystic carcinoma of alveolar mucosa: A rare case report p. 54
Yashwant Ingale, Sushma Bommanavar, Manjusha Ingale, Rajdeep Pavaskar
Adenoid cystic carcinomas are rare epithelial malignancies of salivary glands that show slow growth and local invasion with recurrences seen many years after diagnosis. Upto 50% of tumors occur in intraoral minor salivary glands usually in the hard palate. Tumors on the alveolar mucosa are extremely rare. We present a case of adenoid cystic carcinoma on the right alveolar mucosa in a 35 year old female. The case is been presented for its rarity.
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Sialolipoma of parotid gland in a 1-year-old male child: A case report and review of literature p. 57
Surekha Ulhas Arakeri, Shilpi Banga
Sialolipoma is a recently described rare histological variant of lipoma, characterized by well-demarcated proliferation of mature adipocytes with secondary entrapment of salivary gland elements. Less than forty cases of sialolipoma have been reported in English literature. This tumor has been reported both in major and minor salivary glands, with the parotid gland being the most common site. Age incidence in this tumor varies from 0 month to 84 years. However, the occurrence of this tumor in an infant is very rare. In the published literature, the size of this tumor varied from 1 to 7 cm. In the present case, the tumor size was >7 cm. To the best of our knowledge, congenital sialolipoma >7 cm has been rarely reported earlier. Hence, this case is presented because of its rarity and unusual gross appearance.
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Polymorphous adenocarcinoma of the oral cavity: A skeptical case mimicking lobular carcinoma of breast and gastric carcinoma p. 60
Purnima Vadla, Sankeerthana Pathipaka, Jayakiran Madala, Venkateswara Rao Guttikonda
Polymorphous adenocarcinoma (PAC) is a distinctive salivary gland neoplasm that predominantly occurs in the minor salivary glands. The tumor is characterized by cytological uniformity, morphological diversity, an infiltrative growth pattern and low metastatic potential. It presents as an asymptomatic, slow- growing mass within the oral cavity. This salivary gland tumor is difficult to diagnose both clinically and histopathologically due to its indolent presentation and diverse architectural pattern which includes various microscopic patterns namely- solid, ductal-tubular, cribriform, trabecular and single-file growth. Hence, Immunohistochemistry plays a very important role in diagnosing this tumor. We hereby report a rare case of PAC occurring in a 50yr old female patient presenting with a lesion in right upper posterior part of alveolus extending to the hard palate.
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Adenoid cystic carcinoma of palate: Report of a solid variant p. 65
Anchal Chandraprakash Srivastava, Suresh R Barpande, Jyoti D Bhavthankar, Mandakini S Mandale
Adenoid cystic carcinomas (ADCCs) are uncommon tumors, comprising <1% of all head-and-neck cancers and 20%–25% of all salivary cancers. The most common presenting symptom of the lesion is a slowly growing mass followed by pain due to the propensity of these tumors for perineural invasion. The tumor is most often clinically deceptive by its small size and slow growth, which actually overlies its extensive subclinical invasion and marked ability for early metastasis making the prognosis questionable, aptly referred as “wolf in sheep's clothing.” Cribriform, tubular and solid are the three recognized histopathological patterns. In ADCC, one of the important prognostic factors is the histological grade determined by the percentage of solid component in the tumor, which in the present study comprise more than 30% of the entire lesion rendering it an unfavorable prognosis.
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Keratinizing pleomorphic adenoma: An unusual case report p. 69
Aanchal Tandon, Rohit Jaiswal, Safia Siddiqui, Bharadwaj Bordoloi
Pleomorphic adenoma (PA) is the most common benign tumor of major or minor salivary glands. PA exhibits a great histological diversity, such as differentiation into oncocytic, sebaceous, mucinous, squamous, chondroid, osseous or adipose cells. Squamous metaplasia rarely results in the formation of extensive keratin-filled cyst lined by squamous epithelium. Extensive squamous metaplasia can be mistaken for malignancy, including mucoepidermoid carcinoma and squamous cell carcinoma. Here, we report a case of slowly enlarging PA with extensive squamous metaplasia and keratin cyst formations in a minor salivary gland in hard palate and discuss its microscopic features.
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Juvenile primary extranasopharyngeal angiofibroma, presenting as cheek swelling p. 73
J Nandhini, S Ramasamy, Ronak Nazir Kaul, Ravi David Austin
Angiofibroma is a locally advancing immensely vascular tumor that essentially arises from the nasopharynx. The clinical characteristics of extranasopharyngeal angiofibroma (ENA) do not accord to that of nasopharyngeal angiofibroma and can present a diagnostic confront. We describe a case of primary juvenile ENA in a 19-year-old patient who presented with a rapidly enlarging mass of the cheek region. The case is unusual because of its anatomic location. The diagnostic and management particulars are sketched.
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Leukemic gingival enlargement: A case report and review of literature p. 77
Kanika Chowdhri, Shruti Tandon, Arundeep Kaur Lamba, Farrukh Faraz
The oral cavity manifests signs of various systemic diseases. This entails thorough examination of the oral mucosa, gingiva, teeth, tongue and other oral tissues. Occasionally, oral signs can be an expression of systemic conditions such as endocrine imbalance, nutritional deficiencies and blood disorders. Leukemia is a malignancy of white blood cells, which may result in significant morbidity and mortality. Oral changes maybe the first and only presenting features in leukemia patients, making it imperative for the dentist to diagnose the disease accurately.
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Proliferative endophytic lesion of the maxilla: A diagnostic challenge p. 82
R Shesha Prasad, Aditya Moorthy, Abhishek Bhadranna, Anuradha Pai
Carcinoma cuniculatum (CC) is an extremely rare neoplasm of the oral cavity. It is essentially a well-differentiated variety of squamous cell carcinoma and shows lower metastasis risk. The histological features of CC can mimic that of papillary squamous cell carcinoma or verrucous carcinoma. CC commonly affects the plantar region of the foot, and very few cases have been described in the oral cavity. The classical histological characteristics noted are infiltration of underlying connective tissue by squamous epithelium with keratin cores and keratin-filled crypts. Herewith, we present a case of CC of the right maxilla in a young Dravidian male patient who works in a battery factory. The case presented a diagnostic challenge both clinically and histopathologically before it was completely resected using a total maxillectomy technique. The case delineates the diagnostic challenge and management of the disease.
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Cutaneous horn: A mask to underlying malignancy p. 87
Rashmi GS Phulari, Rajendrasinh Rathore, Trupti Pramod Talegaon, Arpan Shah
Cutaneous horns (cornu cutaneum) are uncommon lesions consisting of keratotic material resembling that of an animal horn. It is a conical- or cylindrical-shaped excessive hyperkeratosis of variable size ranging from few millimeters to several centimeters with a variable in size and shape, such as cylindrical, conical, pointed, transversely or longitudinally corrugated, or curved like a ram's horn. The lesions typically occur in sun-exposed areas, particularly the face, ear, nose, forearms and dorsum of hands. Even though 60% of the cutaneous horns are benign in nature, the possibility of skin cancer should always be kept in mind. The clinical diagnosis includes various benign and malignant lesions at its base. Lesions associated with cutaneous horn are keratosis, sebaceous molluscum, verruca, trichilemmal, Bowen's disease, epidermoid carcinoma, malignant melanoma and basal cell carcinoma. Herewith, we report a case of cutaneous horn on the upper lip vermillion masking the underlying malignancy at its base.
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Benign lymphoepithelial cyst of parotid gland: Review and case report p. 91
Jaya Joshi, Sonalee Shah, Deepak Agarwal, Ankit Khasgiwal
Benign lymphoepithelial cyst (BLC), also known as branchial cyst, is an infrequent lesion usually occurs in the parotid gland or the lateral cervical area including lymph nodes. It occurs due to the process of lymphocyte-induced cystic ductular dilatation and is always diagnosed postoperatively by histopathological examination (HPE). These dysontogenetic lesions are usually found in the lateral neck but can also be located in the salivary glands, mostly in the parotids. A 35-year-old male reported to Government Dental College, Indore, before 3 years, with a soft, nontender, gradually increasing, compressible diffuse swelling involving the left parotid gland for the last 9–10 months of size 3.5 cm × 2.5 cm × 4.5 cm at the time of presentation with normal Stensen's duct and facial nerve function. ELISA test was negative, biochemistry indicated high protein content of aspirated fluid and magnetic resonance imaging showed hypertense cystic fluid in both T1- and T2-weighted images, suggesting high protein or hemorrhage and negating a solid lesion. Superficial parotidectomy was done with nonincidental healing. HPE of excisional tissue revealed it to be BLC.
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Papillary carcinoma of thyroid in a thyroglossal cyst p. 98
Jitendra K Rathod, Swati J Rathod, Vishwas Kadam
Thyroglossal duct cyst (TDC) is the most common congenital anomaly of the thyroid gland and the most common congenital cervical abnormality in childhood. Carcinoma arising from a TDC is rare, which composes only 1% of TDC cases. Here, we are reporting a case of 50-year-old male with a swelling in the upper left neck region which was progressively increasing in size over 5 years. Ultrasonography report showed a large lobulated cystic mass. Internal echoes with calcification and there were no neck nodes. Thyroid gland was normal. In the present study, the Sistrunk procedure was used to excise a TDC. Papillary carcinoma was confirmed with the histopathological examination following the surgery. The patient was treated with radioactive iodide and thyroid suppression therapy was given as adjuvant treatment. The patient has been following for 2 years without any metastasis.
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Renal cell carcinoma metastatic to the maxillary gingiva: A case report and review of the literature p. 102
Daria Vasilyeva, Scott M Peters, Elizabeth M Philipone, Angela J Yoon
Tumor metastasis to the oral cavity is rare and is usually an indication of late-stage disease and poor prognosis. While, there are reports of renal cell carcinoma (RCC) metastatic to oral cavity, vast majority of them are to the jaw. Herein, we present a case of a 78-year-old woman with RCC metastasis limited to the oral soft tissue without any bone involvement. As the lesion solely involved maxillary gingiva, it clinically mimicked that of a pyogenic granuloma, which is a reactive, nonneoplastic condition. This case was further complicated as the patient was unaware of primary cancer and appeared to be in good physical health. Her oral metastasis marked the initial manifestation of an otherwise silent primary renal cancer.
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Oral lymphangiomas – clinical and histopathological relations: An immunohistochemically analyzed case series of varied clinical presentations p. 108
Sumanta Kumar Kolay, Rajkumar Parwani, Sangeeta Wanjari, Pallav Singhal
Lymphangiomas are benign malformations that represent hamartoma of malformed lymphatics and are composed of cystically dilated lymphatic channels which do not communicate or drain into other lymphatic channels or veins leading to accumulation of lymph. They are congenital malformations with rare occurrence in oral cavity within which tongue dorsum is the most affected site. Venous malformations are often difficult to differentiate from lymphatic malformations leading to the development of several lymphatic markers like D2-40 which seems to be useful for distinguishing vascular from lymphatic vessels. The most effective treatment is surgery having good prognosis for most of the patients but depending on their location (neck/tongue) and size (large size) they may cause airway obstruction causing death. Herewith, we present the rare cases of oral lymphangiomas of tongue, palate and buccal mucosa with varied and characteristic clinical presentations.
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Mucormycosis in immunocompetent patient resulting in extensive maxillary sequestration p. 112
Deepak Venkatesh, Satyajit Dandagi, Pramod Redder Chandrappa, KN Hema
Mucormycosis or zygomycosis, also called phycomycosis, is an uncommon, invasive, potentially lethal and an aggressive fungal infection of the order Mucorales that usually affects patients with alteration of their immunological system. From its initial description (Paltauf, 1885), this entity still has a high mortality. Imaging techniques are not usually diagnostic, and cultures are not totally reliable. Definitive diagnosis is exclusively obtained by means of histopathological examination. Early recognition and aggressive treatment are of paramount importance and have reduced the mortality and morbidity. We present here a case report of oral mucormycosis in a 32-year-old male, immunocompetent individual resulting in extensive maxillary sequestration.
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Cervicofacial infection in a 3-year-old child with regional odontodysplasia: A case report and literature review p. 117
Eleanor Bowden, Conor Carroll, Mishaal Gill, Rodney Llewelyn
Regional odontodysplasia (RO) also known as ghost teeth is a rare developmental anomaly affecting the mesodermal and ectodermal components of teeth with characteristic clinical and radiographic findings. The enamel and dentin are hypomineralized and hypocalcified with short roots and open apices. The affected teeth have an abnormal morphology, meaning they are fragile and thin and liable to fracture and decay. Radiographically, there is a marked reduction in radiodensity with little distinction between the enamel and dentin, hence the term “ghost teeth.” RO generally affects one particular segment in one or both arches of the maxilla or mandible and can affect both the primary and permanent dentition. We report a unique case of a 3-year-old female who presented to Alder Hey Children's Hospital acutely unwell with a left-sided cervicofacial swelling from RO affecting the entire portion of the left maxilla.
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Leprosy of the hard palate: A rare case report p. 121
Sushma Bommanavar, Yashwant Ingale, Manjusha Ingale, Sanyukta Ingale
Leprosy is a chronic granulomatous infection caused by Mycobacterium leprae, a bacillus that presents a peculiar tropism for the skin and peripheral nerves. Leprosy instigates various types of clinical presentation and exerts influence on the patient's immune response. The clinical gamut of leprosy ranges from the tuberculoid form (TT) to the disseminative and progressive lepromatous form (LL). Oral lesions are uncommon but, when present, occur in the lepromatous form and are broadly divided into nonspecific and specific lesions. In this article, we present a case of leprosy of the hard palate in a 25-year-old male. The case is been presented for its rarity.
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Tuberculosis of the maxillary sinus masquerading as a facial abscess, a unique occurrence p. 126
Dilip Kumar Rayapati, NT Prashanth, Vinod Rangan, Prithviraj Raj Kalakunta
The incidence of tuberculosis is a frequent finding especially in developing countries owing to a significant population of the people belonging to the lower socioeconomic strata with poor hygiene and nutrition. Tubercular involvement of the paranasal sinuses is usually asymptomatic until it reaches an advanced state and when it involves the maxillary sinus, this initial period of quiescence often mimics the clinical presentation of a chronic sinus infection, which often leads to a diagnostic dilemma. Symptoms commonly reported are chronic sinusitis and rhinorrhea, both of which being nonspecific in nature leading to a quandary. We herewith present a 50-year-old patient who presented with symptoms of an odontogenic infection involving the right maxillary molars. Primary management did not yield to the resolution of presenting symptoms. On biopsy, through an endoscopic approach and subsequent histopathological examination, the diagnosis of tuberculosis was arrived at. The patient responded well to antitubercular drug therapy.
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Fusion of maxillary central incisors with mesiodens p. 131
Madhuram Krishnamurthy, V Naveen Kumar, Ashok Leburu, Nadeem Jeddy
Fusion and gemination are developmental anomalies which are quite similar to each other but can be distinguished from each other if properly assessed. Fusion and gemination have been described as a result of developmental anomalies of dental tissues. The exact etiology is still unknown, but a genetic predisposition is suggested. This article highlights the importance of clinical and radiographic correlation in arriving at a definitive diagnosis.
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Trigeminal neuralgia induced by odontogenic keratocyst associated with impacted supernumerary teeth: A rare case report p. 135
Annaji Araleri Gopalkrishna, BN Tejasree, M Manjunath, DP Ashwin
Odontogenic keratocyst(OKC)is a cyst oftooth origin with an aggressive behavior including a high recurrence rate, it has been rechristened to keratocystic odontogenic tumor(KCOT) as it be the reflects its neoplastic nature. We report a case of KCOT in association with an impacted supernumerary tooth along with Trigeminal Neuralgia, that subsided by itself after removal of the cyst.
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