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    Table of Contents - Current issue
February 2020
Volume 24 | Issue 4 (Supplement)
Page Nos. 1-142

Online since Friday, February 28, 2020

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From the editor's desk p. 1
Smitha Thammaiah
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Papilliferous keratoameloblastoma – A rare entity: A case report with a review of literature Highly accessed article p. 2
Puneeth H Kuberappa, Ananthaneni Anuradha, Mohammad Asif Kiresur, Bhavana S Bagalad
Ameloblastoma is the most common odontogenic tumor which presents with a variety of histopathological patterns. Among all, papilliferous keratoameloblastoma (PKA) is a very rare type which is characterized by multiple epithelial cysts of varying size, which are lined by non-keratinized papilliferous epithelium which is filled with necrotic desquamated epithelial cells. In this study, we reported PKA with characteristic ameloblastic features in a 65-year-old male patient who presented with a swelling in the right mandibular body region. This is the seventh case of PKA to be reported in the English literature till date. Present case showed multicystic areas in incision biopsy which lead to misdiagnosis as calcifying odontogenic cyst with adenomatoid odontogenic tumor, but in excision biopsy which turned out to be papilliferous keratoameloblastoma, further in this paper we had discussed all the areas which lead to misdiagnosis of calcifying odontogenic cyst with adenomatoid odontogenic tumor. In outlook, more cases will put an insight to the behavioral aspects of this rare histological type of ameloblastoma.
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Is odontogenic keratocyst an endodontic enigma? A rare case report of management of odontogenic keratocyst in anterior mandible p. 7
BR Prashanth, Mythreyee S Vidhya, Rupali Karale, G Vinay Kumar
Odontogenic keratocyst (OKC), a rare, locally aggressive developmental cyst, is found incidentally on dental radiographs, most commonly in the posterior mandible. When it occurs in the periapical region in association with nonresponding teeth to pulp sensibility tests, it is often misdiagnosed as other endodontic lesions such as radicular cyst, lateral periodontal cyst and dentigerous cyst. This case report describes the rare occurrence of OKC in the anterior mandibular region managed successfully with endodontic treatment and re-interventional surgery in a conservative approach. This case report also emphasizes on the importance of histopathological examination of the surgically excised tissue specimens as the clinical, radiographic and histopathological correlations are essential for successful treatment.
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Benign cementoblastoma associated with primary mandibular second molar: A rare case report p. 11
Mallayya C Hiremath, SK Srinath, Sahana Srinath, T Ashwathy
Cementoblastoma is a rare benign odontogenic neoplasm of the jaws commonly occurring in the young age group. Here, we present a case of swelling associated with mandibular primary second molar. A provisional diagnosis of cementoblastoma was made based on the clinical and radiographic features. The lesion was surgically removed along with the associated primary mandibular second molar. Histopathological examination confirmed it as benign cementoblastoma. One-year follow-up was done, the healing was satisfactory and no signs of recurrence were observed.
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Focal cemento-osseous dysplasia: A case report with a review of literature p. 15
Anuprita S Salvi, Sangeeta Patankar, Khushal Desai, Divyesh Wankhedkar
Cemento-osseous dysplasia (COD) is the most prevalent lesion and presents as an asymptomatic, mixed radiolucent/radiopaque lesion in the tooth-bearing region of the jawbones. Histological features of COD include a cellular connective tissue stroma interspersed by the islands of woven or lamellar bone and cementum-like calcifications. Radiographically, the early COD lesions appear radiolucent, whereas in the mature lesions, radiopacities are observed surrounded by a thin rim of radiolucency. Early lesions tend to get misdiagnosed as their radiographic appearance mimics periapical cyst or granuloma. In the mixed radiolucent-radiopaque stage, the lesion could be confused with chronic sclerosing osteomyelitis, cemento-ossifying fibroma, odontoma and osteoblastoma. A correct diagnosis is of utmost importance as most of the CODs are self-limiting and nonneoplastic and do not require surgical intervention. However, periodic follow-up is recommended because occasional cases of focal COD are known to progress into florid COD. Here, we present the case of focal COD in a 27-year-old male patient.
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Focal cemento-osseous dysplasia p. 19
S Shamala Ravikumar, G Vasupradha, TR Menaka, S Pradeep Sankar
Focal cemento-osseous dysplasia (FCOD) is a benign fibro-osseous lesion of bone characterized by the replacement of normal bone by fibrous tissue and subsequently followed by its calcification with osseous and cementum-like material. It is mostly asymptomatic in nature and requires no treatment. When secondarily infected, it becomes symptomatic and intervention is required. Here, we report a case of symptomatic FCOD of mandible in a 52-year-old female patient. Histopathological evaluation of the biopsy specimen by ground sections and decalcified sections aided in the final diagnosis of the case.
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A distinct fibro-osseous lesion of the jaws affecting the maxilla p. 23
Wilber Edison Bernaola-Paredes, Norberto Nobuo Sugaya, Mariana Lobo Bergamini, Paulo Henrique Braz-Silva
The differential diagnosis of fibro-osseous lesions (FOL) presents oral clinician stomatologists with a challenging task because of their similar clinical, radiographic and histopathological behavior that will also hinder the therapeutic planning. Presentation of case to describe the clinical, radiographic and histopathological features of an unusual case of FOL presented by the patient, a Black woman, and the results obtained with the conservative surgical treatment and 3-month follow-up. The presence of cementum in the histopathological analysis of cemento-osseous dysplasias, according to the current literature, is an important factor for the diagnosis of this lesion. Considering the characteristics presented by this particular case, we could suggest another entity that could be named a benign cemento-osseous lesion with adult onset. Some occurrences, as in the present case, do not fit properly into the current World Health Organization classification, thereby generating some doubts concerning the correct management of these patients. Conservative surgical treatment is the first choice in the management of these lesions due to their self-limiting nature, which show a behavior of clinical involution.
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Recurrent adenoid cystic carcinoma of paranasal sinuses: A rare case report p. 28
Sarah Abdul Wahed, Katakam Pradeepthi, Madala Jaya Kiran, Guttikonda Venkateswara Rao
Adenoid cystic carcinoma (ACC) is a distinct salivary gland neoplasm that predominantly occurs in the minor salivary gland. ACC is an uncommon tumor that progress slowly affecting 5% of paranasal sinuses with high distant metastasis rate in advance stages. This salivary gland tumor is difficult to diagnose both clinically and histopathologically due to its indolent presentation and diverse histopathological patterns. Hence, immunohistochemistry plays a decisive role in diagnosing this tumor. A recurrent case of ACC of paranasal sinuses in the maxillary sinus of a 41-year-old female patient without any extraoral abnormality is reported.
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Benign lymphoepithelial lesion of the minor salivary gland – A rare presentation as a palatal swelling p. 33
Chandrasekaran Krithika, J Sreedevi, B Sivapathasundharam, VR Nithya
Benign lymphoepithelial lesion (BLEL) is characterized by extensive lymphocytic infiltration of the major salivary glands and may be associated with Sjogren's syndrome or HIV infection. The involvement of the palatal minor salivary glands is extremely rare. We report an isolated case of BLEL affecting the palatal minor salivary glands, presenting as a palatal swelling in a 37-year-old female patient. Serological tests ruled out potential comorbid conditions. Cone-beam computed tomography showed a palatal soft-tissue mass with thinning of the adjacent cortical plates. A histopathological examination revealed salivary gland tissue with significant acinar destruction, dense lymphocytic infiltration and focal myoepithelial islands. Therefore, BLEL may be considered as a rare differential diagnostic possibility of a palatal soft-tissue mass lesion.
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Intraosseous adenoid cystic carcinoma of the mandible: A rare presentation p. 37
Sudip Indu, Indranil Deb Roy
Central adenoid cystic carcinoma (ACC) of the mandible is a rare entity arising from minor salivary glands. Their clinical and radiographic features may be similar to any odontogenic/nonodontogenic pathology, thus making their precise diagnosis wearisome. ACC is well documented for its protracted clinical course, perineural invasion, multiple recurrence rates and its propensity for distant metastases. The aim of this study is to report a rare case of primary central ACC of the mandible with an unusual presentation in terms of location, however, demonstrating the classical features of ACC in histopathology and positivity in immunohistochemistry to S100, calponin and CD117. A thorough clinical, radiographic, CT evaluation and meticulous metastatic workup along with long-term follow-up is advised in such cases. Although central ACC is extremely rare, especially in the anterior mandible, it should be included in the differentials for lesions of the mandible.
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Primary intraosseous adenoid cystic carcinoma of the mandible: A rare clinical entity p. 42
Vandana Reddy, Vijay Wadhwan, KV Arunkumar, Arvind Venkatesh
Central intraosseous adenoid cystic carcinoma (ACC) of the mandible, formerly known as cylindroma, is a rare neoplasm with only 47 cases reported in the literature. We present a case of central ACC involving the mandible of a 55-year-old male patient.
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A miscellany of cribriform pattern, squamous metaplasia and clear cells in pleomorphic adenoma of upper lip: A diagnostic paradox p. 46
Sayani Shome, Neha Shah, SK Abdul Mahmud, Mousumi Pal
Pleomorphic Adenoma (PA), a benign neoplasm of glandular origin most commonly involves major salivary glands. It is rare in minor salivary glands such as hard palate, upper lip and buccal mucosa, frequently affecting middle aged females. PA comprises diverse histopathologic features of epithelial, myoepithelial and mesenchymal components. Aberrant histopathologic features in Pleomorhic Adenoma thus calls for judicious discrimination from alike entities which facilitates appropriate surgical management. Here we present a case report of PA in upper lip in a 25 year old female patient showing uncommon findings like clear cells, squamous metaplasia and cribriform pattern.
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Infantile anterior maxillary swelling: A diagnostician's dilemma p. 51
Dharamraj B Patil, Vijay Wadhwan, Sheetal D Patil
Anterior maxillary swellings are commonly encountered in the adolescents and adults and they represent lesions ranging from cysts to tumors which can be both benign as well as malignant. However the anterior maxillary swellings are a rare phenomenon in the infants and toddlers and they generally are indicative of an aggressive lesion. We hereby present a case of a rapidly growing infantile swelling which was histopathologically diagnosed as Peripheral Giant Cell Granuloma.
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Oral medullary plasmacytoma: Rare case reports p. 55
Malathi Narasimhan, Ravindran Chinnaswamy, GV V Giri, Vijaya Nirmala Subramani
Medullary plasmacytoma (MP) or osseous or solitary bone plasmacytoma is a specializing hematopathology lesion typically present with local symptoms, such as pain, paresthesia and pathologic bone fractures as a result of proliferation of plasma cells. The most often involved sites are active hematopoietic long bones and the vertebrae. The clinical course of disease is identical to spectrum of other plasma cell dyscrasias. The diagnostic criteria include punched-out radiolucencies, monoclonal plasma cells and M protein. This lesion should be considered for the differential diagnosis of bone tumors. It is highly radiosensitive although combination modalities of radiation, surgery and chemotherapy have been used in the treatment. The long-term follow-up is essential. We report two rare cases of oral MP with unusual clinical presentation.
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Multifocal osteolytic lesions of jaw as a road map to diagnosis of brown tumor of hyperparathyroidism: A rare case report with review of literature p. 59
Suchitra Gosavi, Harpreet Kaur, Pramod Gandhi
Brown tumor is unifocal or multifocal bone disease which represents terminal stage of hyperparathyroidism (HPT)-dependent bone pathology. It is recognized as a component of metabolic bone disease called osteitis fibrosa cystica generalisata or Von Recklinghausen disease of bone. HPT was first described by Von Recklinghausen in 1891. Brown tumor diagnosis nowadays is less frequently encountered because of early stage detection of HPT. This early detection is possible due to routine blood screening in asymptomatic adults or during evaluation of osteoporosis. Histologically, it may resemble any other giant cell lesion of the jaw that imposes diagnostic challenge and delay in treatment. We are introducing a case report of a 30-year-old female patient presented with multifocal osteolytic lesions in mandible with histopathology depictive of giant cell granuloma. Further biochemical investigations and X-ray skeletal changes raised the suspicion of primary HPT which was confirmed by parathyroid scintigraphy revealing parathyroid adenoma. The main purpose of this case report is to reinforce the role of oral examination in diagnosis of systemic diseases and to propose a diagnostic layout/algorithm when giant cells are present in biopsy specimen. Review of literature showing brown tumor of oral cavity associated with PHPT is discussed.
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Giant cell-rich osteosarcoma – A rare case p. 67
Aishika Mallick, Neha Shah, SK Abdul Mahmud, Sanjeet Kumar Das
Giant cell-rich osteosarcoma (GCRO) is an exceedingly rare histological variant of conventional primary osteosarcoma. It constitutes about 1%–3% of all osteosarcomas, and is extremely uncommon in the maxillofacial region. The unusual histopathological appearance and the rarity of the lesion poses a great diagnostic challenge. This article aims to present a rare case of GCRO involving the mandible in a 52-year-old male patient.
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A rare presentation of benign fibrous histiocytoma in the maxilla p. 73
Aishwariya Mohanty, Pallavi Mishra, Harish Kumar, Abikshyeet Panda
Fibrous histiocytoma is a soft-tissue tumor that may present as a fibrous mass anywhere in the human body. The involvement of the oral cavity is rare. We here report a case of benign fibrous histiocytoma localized in the maxilla. A 25-year-old male presented with a slowly increasing large painless mass over the left side of the upper jaw for 5 months. The swelling was of gradual onset, slowly progressive and was not associated with pain, tenderness, or discharge. The radiographic appearance showed a multilocular mixed radiodense, radiolucent lesion in the left posterior maxilla. The histopathological report showed connective tissue component having streaming fascicles of spindle-shaped cells showing a storiform pattern. Many areas showed foamy histiocytes along with few multinucleated giant cells. The cells were appearing benign without any appearance of atypia. The tumor cells were positive for CD-68 and vimentin and negative for CD-34 and S100. The lesion was excised under general anesthesia.
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Diffused mixed B-cell non-Hodgkin lymphoma of mandible p. 77
Asha Karadwal, Shailja Chatterjee, Kush Pathak, Robin Sabharwal
Lymphomas are the third-most common cancer of the oral cavity after squamous cell carcinomas and salivary gland tumors. It is characterized by proliferation of lymphoid cells and their precursor. Diffuse B-cell non-Hodgkin lymphoma is the most common histological type of lymphoma in the head-and-neck region and most commonly affects older men in their seventh decade of life.
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Oral malignant melanoma p. 82
Sahana Ashok, Srikanth Damera, Siva Ganesh, Rojalakshmi Karri
Malignant melanoma in the head and neck area is rare. The incidence of oral malignant melanomas of the head and neck is approximately four per 10 million populations per year. They are derived from epidermal melanocytes and are most frequently seen on the palate and maxillary gingiva. They are asymptomatic initially but become painful with progress and enlargement. Later, they are associated with ulceration, bleeding, mobility of tooth, paresthesia and ill-fitting prosthesis. The diagnosis is often delayed due to silent growth and development of the lesion. Oral malignant melanomas are associated with poor prognosis due to their invasive and metastasizing tendencies. This case report is presented to emphasize the role of a dentist in identifying the pigmented lesions of the oral cavity.
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Malignant peripheral nerve sheath tumor: A rare malignancy p. 86
Sheetal Sharma, Jigna S Shah, Harleen Bali
Malignant peripheral nerve sheath tumor (MPNST) is also termed as spindle cell malignancy of the peripheral nerve Schwann cell. It is a rare and highly aggressive, soft-tissue sarcoma of ectomesenchymal origin that accounts for 10% of all sarcomas and only 10%–12% of all lesions occur in the head-and-neck region, thus making it a rare entity. It arises de novo or from the preexisting benign neurofibroma. The diagnosis of MPNST is one of the most elusive among the soft-tissue tumors because of its greater variability in overall presentation both clinically and histologically. This difficulty can be overcome by the use of immunohistochemistry. This article presents a rare case of MPNST of the oral cavity in a 40-year-old female patient with a brief review of the current literature.
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Intraoral plasmablastic lymphoma as a primary oral manifestation: A case report and review of literature p. 91
Revati Deshmukh, Purva Abhyankar, Amit Mhapuskar, Harshal Varpe
Plasmablastic lymphoma (PBL) is an aggressive type of large B-cell lymphoma as stated in the WHO classification of 2008. It is a rare form of non-Hodgkin's lymphoma, generally seen in human immunodeficiency virus (HIV)-acquired immunodeficiency syndrome affected individuals. A case of a 42-year-old female patient is presented here. The patient complained of swelling in the lower right back tooth region and presented with a history of extraction of molars. The underlying HIV status was detected after the oral examination. The diagnosis of PBL was confirmed with immunohistochemical analysis.
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Benign fibrous histiocytoma of the lower lip p. 97
Nagaraja Anand, Rashmeet Kaur, Sujata Saxena, Nandini Bhardwaj
The fibrous histiocytoma is a soft-tissue neoplasm of the biphasic cell population of fibroblasts and histiocytes that affects the dermis and the subcutaneous tissue. The objective of this article is to report a case of benign fibrous histiocytoma (BFH) of the lower lip in a 32-year-old female patient with a chief complaint of swelling in the lower lip for the past 1 month. With diagnostic clinical hypothesis of fibrous hyperplasia, fibrous histiocytoma and mucocele, an excisional biopsy was performed. The histopathological examination revealed a nonencapsulated proliferation of spindle cells with some giant multinucleated cells in the periphery of the lesion. Multinucleated giant cells and lymphocytes were noted throughout the lesion. Immunohistochemical reactions were performed, staining only CD68 in the multinucleated giant cells. According to these characteristics, the final diagnosis was BFH.
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Abrikossoff's tumor of the tongue: Report of three cases and review of the literature p. 101
Ida Barca, Raffaella Cordaro, Amerigo Giudice, Maria Giulia Cristofaro
Abrikossoff 's tumor, also called granular cell tumor (GCT), is a rare benign neoplasm of the soft tissues. In almost 70% of the cases, it occurs in the head-and-neck region (especially in the tongue), even though it may present in every other part of the body. This neoplasm has a benign behavior usually, but there have been described a malignant transformation in 2%–3% of the cases. The characterization of this tumor depends on its clinical and histopathological findings. The purpose of our work is to report three uncommon cases of oral Abrikossoff's tumor located on the tongue in young patients referred to our department of maxillofacial surgery of “University Magna Graecia” in Catanzaro, also reviewing of the literature.
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A rare case report of neurofibromatosis type 1 in a 12-year-old child: A 15-month follow-up p. 106
Srinivas Nallanchakrava, Manoj Kumar Mallela, V Shiva Kumar Jeenepalli, HM Niharika
Neurofibromatosis type 1 (NF-1) or von Recklinghausen's disease is a rare genetic disorder characterized by the development of multiple noncancerous (benign) tumors of nerves and skin (neurofibromas). Head-and-neck neurofibroma is generally located in the soft tissue. Here, we present a case of a 12-year-old girl with NF-1. The disease started in childhood with the appearance of multiple hyperpigmented skin macules. The girl presents generalized freckling and café au lait spots throughout the body and a diffused swelling measuring about 4 cm × 3 cm, extending from the right maxillary hard palate region to the midpalate. The diagnosis of NF-1 was made according to the presence of two or more diagnostic criteria of the National Institute of Health Consensus Development Conference. No recurrence was observed in a 15-month follow-up after extensive surgical ablation.
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Adenosquamous carcinoma of the tongue: A case report and an overview of histogenetic concepts p. 110
Manoj Prabhakar, B Sabarinath, B Sivapathasundharam, V Vasanthakumar
Adenosquamous carcinoma (ASC), a rare variant of squamous cell carcinoma, is an aggressive and highly infiltrating epithelial neoplasm, which has a distinct histomorphological features comprising simultaneous areas of squamous cell carcinoma and adenocarcinoma. Some researchers consider it as a controversial neoplasm, as it may arise from the ducts of the minor salivary gland or from the overlying surface epithelium. In the head-and-neck region, commonly affected sites include paranasal sinuses, larynx and oral cavity. Areas of occurrence in the oral cavity include palate, tonsillar pillar areas, tongue and floor of the mouth. The prognosis of the neoplasm is considered poor due to its early recurrence, local and distant metastasis and low survival rate. This article reports a case of ASC affecting the posterior part of the tongue in a 50-year-old male patient and an overview of its histogenetic concepts.
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Adipocytic tumors of orofacial region: Clinicopathologic appraisal of ten cases with a review of its variants p. 115
Nivedita Kaorey, Mandakini Mandale, Jyoti Bhavthankar
Lipomas are the most common benign tumors. However, their presence in oral cavity is albeit erratic. Even scarcer is the prevalence of the various histopathological variants. We, hereby, report the wide range of clinical and histopathologic presentations of these uncommon entities affecting the orofacial region. Discussed herein are six cases of lipomas, two cases of intramuscular lipomas and one case each of osteolipoma and sialolipoma.
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Monoradicular primary mandibular first molar: A rare case in Aseer Province of Saudi Arabia p. 120
Zainah M Al-Shahrani, Usha Balan, Khalil Ibrahim Assiri, Abdulaziz M Maken Al Qarni
Morphological variations can occur in primary and permanent teeth. Genetic and environmental factors could be responsible for various dental anomalies. Anomalous teeth are usually asymptomatic and are diagnosed during routine oral examination. These anomalies may increase caries susceptibility and complicate dental treatment procedures such as extraction or root canal treatment. We report a rare case of mandibular first molar with a single root and a root canal.
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Nasofacial rhinoentomophthoramycosis: A report of two cases from Central India p. 124
Archana B Wankhade, Priyadarshini Patro, Ripu Daman Arora, Nitin M Nagarkar
Rhinoentomophthoromycosis due to Conidiobolus coronatus is a rare, chronic, granulomatous disease, occurring mainly in tropical countries including India. We report two cases of rhinoentomophthoromycosis in an 18-year-old female and a farmer of 35 years residents of Chhattisgarh shifted from Madhya Pradesh and Orrisa. It was diagnosed by microscopy and isolation C. coronatus in culture. The patient presented with a swollen nose with obstruction that had progressed slowly over 1 year. His nasal swelling was bilateral, diffuse, mildly tender, erythematous, nonpitting, with mucosal crusting and hypertrophy of inferior turbinates but no regional lymphadenopathy. Culture of tissue from the nasal biopsy on sabouraud dextrose agar yielded multiple colonies of a mold with satellite smaller colonies at periphery. The isolate demonstrated the macroscopic and microscopic morphologic characteristics of C. coronatus. The patients were earlier treated with itraconazole or its combination with potassium iodide and the patients were treated successfully with amphotericin B.
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Oral lichen planus in an 8-year-old child: A case report with a brief literature review p. 128
Shamimul Hasan, Shahnaz Mansoori, Mohd Irfan Ansari, Safia Siddiqui
Lichen planus (LP) is a chronic autoimmune condition of uncertain etiopathogenesis and usually affects the skin, oro-genital mucosa, nail and scalp appendages. LP is primarily seen in middle-aged individuals, and oral lesions of LP in children are relatively uncommon. Herewith, we report a case of oral LP in an 8-year-old boy, which regressed well with the treatment modality.
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Immunoglobulin G4-related chronic sclerosing sialadenitis: An emerging entity p. 135
Sushama Govindrao Gurwale, Charusheela Rajesh Gore, Ishita Gulati, Indranil Dey
Chronic sclerosing sialadenitis is associated with the immunoglobulin G4 (IgG4)-related disease (RD) spectrum. IgG4-RD is a newly recognized immunomediated fibroinflammatory condition characterized by several features: a tendency to form tumefactive lesions at multiple sites, lymphoplasmacytic infiltrate, fibrosis and obliterative phlebitis. Often but not always, the serum IgG4 concentrations are also elevated. Immunohistochemistry for IgG4 is helpful to clinch the diagnosis. Here, we describe a case of 65-year-old male with IgG4-related chronic sclerosing sialadenitis of the s ubmandibular gland. We have discussed the histopathological criteria to diagnose this entity.
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Fine needle cytology features of an atypical presentation of infectious mononucleosis Highly accessed article p. 139
Roberto Gerber-Mora, Verónica Peña y Lillo, Ricardo Moreno-Silva, Wilfredo González-Arriagada
Infectious mononucleosis (IM) is a very common disease, and although in most instances, the patient develops an asymptomatic infection, other patients progress into an array of signs and symptoms that tend to be characteristic of the pathological process, guiding the clinician into choosing the right laboratory examinations under the suspicion of this illness. The most common symptoms are pharyngitis, lymphadenopathies and pyrexia, and the probabilities to develop this mononucleosis triad become greater with age. In other instances, IM can be a challenging disease to diagnose, mainly because the patient debuts with unusual symptoms such as nausea, skin eruptions, diarrhea and epigastric discomfort. The aim of the present article is to report the case of a 21-year-old female with a reactivation of IM, developing only multiple, asymptomatic lymph node enlargements in the head and neck region, showing no other sign or symptom.
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