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    Table of Contents - Current issue
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February Supplement 2023
Volume 27 | Issue 5 (Supplement)
Page Nos. 1-108

Online since Saturday, February 4, 2023

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CASE REPORTS  

Non-familial paediatric oral myofibroma - A masquerade. Case report with a brief literature review Highly accessed article p. 1
Spoorti Kulkarni, Adarsh Kudva, Srikanth Gadicherla, Sunitha Carnelio
DOI:10.4103/jomfp.jomfp_141_22  
Myofibromas are fibrous tumours that could be of familial or non-familial origin, belonging to the fibroblastic and myofibroblastic subset with a wide spectrum of clinical behaviour. Oral myofibromas present with a broad range of differential diagnoses, including benign and malignant lesions. Histopathologically, these lesions may imitate many other soft tissue tumours of the oral cavity, such as spindle cell tumours of nerve, smooth muscle cell origin, and other myofibroblastic lesions, thus leading to misdiagnosis and mistreatment. In the present paper, we report a soft tissue lesion, which presented as a growth on the gingivobuccal sulcus in a 7-year-old child. We also emphasise the various differential diagnoses that need to be eliminated and the importance of immunohistochemistry to know the nature of tumour cells in establishing the accurate diagnosis.
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Extensive tongue lymphangioma followed for eight years: Case report Highly accessed article p. 6
Francisco Nunes Junior, Everton J da Silva, Ana L. L. M Paz, Luiz E. R Volpato
DOI:10.4103/jomfp.jomfp_511_20  
Lymphangiomas are benign hamartomatous tumours similar to lymphatic vessel neoplasms, originating from lymphatic tissue sequestration and may or may not communicate with the rest of the system. There are several treatment options for lymphangioma, such as surgery, sclerotherapy, cryotherapy, lasers, steroids and bleomycin. Although surgery is the most indicated treatment, it can result in severe sequelae and loss of function in patients. The present case shows a 5-year-old patient diagnosed with extensive tongue lymphangioma and, due to the mutilation that would be caused by its complete excision, the option was for more conservative treatments such as laser therapy and cryotherapy. After follow-up for 8 years, the patient presents with preserved functions and controlled lesion.
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Low-grade myofibroblastic sarcoma of the mandible: A rare tumour of childhood Highly accessed article p. 10
Saikat Mitra, Satnam S Jolly, Anindita Sinha, Debajyoti Chatterjee
DOI:10.4103/jomfp.jomfp_256_22  
Low-grade myofibroblastic sarcoma is a rare and indolent tumour of soft tissue. This tumour is relatively common in the head and neck region followed by extremities. Primary low-grade myofibroblastic sarcoma of the mandible is very rarely reported and the occurrence of this tumour in a child is very unusual. A 7-year-old male child presented with a swelling in right angle of mandible. X-ray and computed tomography scan showed a lytic lesion. The lesion was excised and the tissue was sent for histopathological evaluation, which revealed a cellular spindle cell neoplasm arranged in fascicles. The tumour was partly circumscribed and lobulated. On immunohistochemistry (IHC), these tumour cells showed cytoplasmic positivity for vimentin, and smooth muscle actin showed 'tram-track' pattern of positivity. The case was diagnosed as low-grade myofibroblastic sarcoma. There are no definite clinical features or pathognomonic radiological appearances of this tumour that can differentiate this rare tumour from other commonly encountered gnathic bone tumours, such as osteosarcoma, inflammatory myofibroblastic tumour, etc., Histopathological diagnosis coupled with ancillary investigations such as IHC is important to establish a definite diagnosis and rule out the differentials. The exact biological behaviour of this tumour is not known.
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Glomus tumor in the buccal mucosa: A case report and review of the literature p. 15
Bahar Afroozi, Fahimeh Rezazadeh, Zohreh Jaafari-Ashkavandi, Saeid Tavanafar
DOI:10.4103/jomfp.jomfp_232_22  
Glomus tumors are rare benign neoplasms usually occurring in the upper and lower extremities. However, oral cavity involvement is rare, with only a few case reports. We present a 37-year-old male patient with a chief complaint of an exophytic lesion in the left buccal mucosa for one year referred to our center. At the time, he did not have any pain or lymphadenopathy. The patient underwent surgery using a high-power CO2 laser. His histopathological examination revealed a glomus tumor. After 2 years of follow-up, no evidence of recurrence was detected. Glomus tumors should be taken seriously when patients have a painless exophytic bluish lesion in the buccal mucosa.
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Oral hibernoma along with multiple lipomas p. 20
Mahaboob Shahnaz, Nair Shalini, Abdul Qayyum Shereefa, Kuruvilla Vikas
DOI:10.4103/jomfp.jomfp_260_22  
'Hibernoma' is a neoplasm that arises from vestiges of fetal brown fat, and its occurrence in oral cavity is extremely rare. Its most common locations include thighs, the inter-scapular region, and the cervical region. In the present case, a 37-year-old male patient reported to our department with a localized swelling on his lower left labial mucosa along with multiple cutaneous well-defined swellings on his right arm and abdominal region. Incisional biopsy was carried out. Histopathological examination revealed sheets of multi-vacuolated eosinophilic cells with the granular cytoplasm interspersed with fat cells suggestive of oral hibernoma. These are rare lesions and could be often a missed-out diagnosis. Therefore, it is imperative to consider oral hibernoma among the commonly considered differential diagnosis of oral mucosal swellings.
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Benign scalp lesion: An unusual presentation of B cell- lymphoblastic lymphoma – A case report p. 24
Ranjini Kudva, Sadaf Khan
DOI:10.4103/jomfp.jomfp_278_21  
This case report highlights the occurrence of B- cell lymphoblastic lymphoma (B-LBL) as a solitary cutaneous lesion without an existing systemic involvement and should be kept in the differentials while dealing with cases presenting with a similar clinical picture. We report the case of a 13-year-old girl who presented with a painful, progressively enlarging swelling in right zygomatico-temporal region, clinically simulated a deep fungal infection/ granulomatous lesion and turned out to be a case of B-LBL without any systemic involvement on further work up. This case is being reported to emphasize that B-LBL should be considered as a differential for an otherwise benign appearing persistent lesion in the head and neck region.
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Intraductal carcinoma of right parapharyngeal space presenting as a fluctuant swelling of retromolar region: Case emphasizing on the histological differential diagnosis p. 28
Deepak Pandiar, Pratibha Ramani, Murugesan Krishnan, Reshma P Krishnan
DOI:10.4103/jomfp.jomfp_288_22  
Intraductal carcinoma (IDC) of salivary gland is an extremely rare malignancy affecting mainly the parotid glands. Intraoral occurrence is seen mainly on the palate where the tumour arises from the minor salivary glands. No previous case has been described in parapharyngeal space. We report a case of low-grade IDC of the anterior compartment of the right parapharyngeal space that clinically resembled a lesion of vascular origin. Due to the extreme rarity of intraductal carcinoma, it may not be considered by dentists and dental specialists in clinical differential diagnoses, leading to delay in treatment. Intraductal carcinoma must be differentiated from its close histological but high-grade mimickers to avoid unnecessary overtreatment and better patient outcome.
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Undifferentiated pleomorphic sarcoma of the floor of mouth: A rare case p. 33
Anjali Sharma, Praveena Vyas, Deshant Agarwal
DOI:10.4103/jomfp.jomfp_107_21  
Undifferentiated pleomorphic sarcoma (UPS) previously called as malignant fibrous histiocytoma comprises a group of high-grade pleomorphic sarcomas that cannot be otherwise classified and considered as a diagnosis of exclusion. In the head neck region, UPS is extremely rare and accounts for 3% of all the undifferentiated pleomorphic sarcomas. Some of the reported sites include maxilla, mandible, buccal mucosa, temperomandibular fossa, tongue, gingiva, paranasal sinuses, salivary glands, and retro-orbital soft tissue. Undifferentiated pleomorphic sarcoma of the floor of the mouth is very rare. To our knowledge, only one case has been reported earlier. We report the second case of undifferentiated pleomorphic sarcoma of the floor of mouth.
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Extra adrenal paragnglioma of oral cavity: A rare case report p. 38
Manas Bajpai, Pradkhshana Vijay, Nilesh Pardhe,
DOI:10.4103/jomfp.jomfp_432_21  
Paragangliomas are neuroendocrine tumors similar to pheochromocytomas but arising from extra adrenal site. It is a very rare tumor in an intraoral site, we found a single case of intraoral paraganglioma in a literature, and hence as per our best knowledge this is only the second case of paraganglioma presented in an intraoral location. We present here a case report of paraganglioma on the ventral surface of the tongue. A 37 years old lady presented with a large asymptomatic swelling on the ventral surface of the tongue from one year. The past medical history and familial history were non relevant to the swelling. Surgical excision was performed and histopathological examination of the specimen revealed the diagnosis of extra adrenal paraganglioma. The case is being presented here for its rarity in an intraoral location.
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Oncocytoma of the parotid gland: A rare benign tumour p. 41
Jaya Singh, Akhilesh Chandra, Tumpuri Srilatha, Trupti Jain, Dinesh Raja, Rahul Agrawal
DOI:10.4103/jomfp.jomfp_437_21  
Oncocytomas are one of the infrequent neoplasms seen in the oral cavity accounting for less than 2% of all neoplasms in the oral cavity with less than 1% chance of malignant transformation. They affect the major salivary glands and have a female predilection. The cognisance of the unique clinical and histopathological features is very important to conclude a confirmatory diagnosis. This paper reviews a case of oncocytoma presented in our department and also elucidates the diagnostic criteria for the same.
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Actinomycosis of parotid gland after maxillary molar extraction, case report and review of literature p. 45
İrem Guvendir, Murat H Karabulut, Itır Ebru Zemheri
DOI:10.4103/jomfp.jomfp_257_22  
Although actinomycosis is frequently seen in the cervicofacial region, it is very rare in the parotid gland. Furthermore, it can be confused with salivary gland malignancies in imaging. The most important underlying causes are cervicofacial trauma, tooth extraction history and poor oral hygiene. A 43-year-old male patient was admitted to otorhinolaryngology department with a complaint of progressive swelling on his cheek. The histopathological examination of fine-needle aspiration (FNA) biopsy showed acute suppurative polymorphous leukocytes and actinomycese hyphae balls within histiocytes. The patient was started on amoxicillin 2 gm per day for two months. With antibiotherapy, the swelling in the patient's parotid gland decreased and completely healed at the end of six months. Primary parotid actinomycosis has been rarely reported in the literature, differential diagnosis is usually problematic as both clinical and radiological findings may mimic parotid tumour; however, it can be useful to apply FNA cytology for a quick diagnosis and treatment.
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Basal cell adenoma of the floor of the mouth masquerading as ranula- A rare case in an unusual location p. 52
Soumeen N Gadkari, Shirin Harnekar, Aarti M Mahajan, Nilima Prakash
DOI:10.4103/jomfp.jomfp_356_22  
A 65-year-old female patient with a history of wearing a denture for 15 years reported with a complaint of pain in the floor of the mouth. On examination, a swelling measuring about 2.2 X1.2 cms was noted in the floor of the mouth on the left side. The lesion was excised under local anesthesia. Histopathology revealed a cystic lesion with basaloid cells arranged in tubules, nests and cribriform pattern. A diagnosis of basal cell adenoma was made and confirmed by immunohistochemistry. We report a rare case of Basal Cell Adenoma of the floor of the mouth which mimicked a ranula. The predominantly cystic nature of this basaloid tumor posed a diagnostic challenge. Histopathological and immunohistochemical analysis to arrive at a definitive diagnosis aid in treatment planning and prognostication.
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Frameshift mutation in exon 17 of PTCH1 gene in Nevoid basal cell carcinoma syndrome: A case report p. 56
Thara Aravind, Vindhya Savithri, Rakesh Suresh, Pramod Subash
DOI:10.4103/jomfp.jomfp_463_20  
Nevoid basal cell carcinoma syndrome (NBCCS) is a rare autosomal dominant disorder characterized by a wide range of developmental abnormalities and a predisposition to neoplasms. In majority of the cases, the presence of multiple and recurrent jaw cysts especially during the first two decades of life is one of the first symptoms of this syndrome. We present here a case of 14-year-old female patient who reported with a chief complaint of facial swelling for 3 weeks. The radiographs revealed multiple cysts in maxilla and mandible. Incisional biopsy of the lesions was done and the histopathologic features were suggestive of odontogenic keratocyst. Further investigations revealed the presence of falx cerebri calcifications and multiple nevi on palms and feet. Genetic study was done to confirm the diagnosis of NBCCS, which showed mutations in PTCH gene. This case stresses the importance of genetic study in suspected cases of NBCCS especially in young patients of nonsyndromic parents.
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Ameloblastic fibroma: A case report p. 60
Abdullah Nasir, Abhisheik Khare, Iqbal Ali, Mohammad I Khan
DOI:10.4103/jomfp.jomfp_337_22  
Ameloblastic fibroma is a rare mixed odontogenic benign tumor that can occur in either mandible or maxilla but mostly it is found in posterior region of mandible. It can present either peripherally or centrally with a majority of the cases predominantly occurring in first two decades of life and mostly affects male patients. It is characterized by epithelial islands and cords submerged in ectomesenchyme that bear resemblance to the dental papilla and enamel organ but without actual hard tissue formation. Ameloblastic fibroma is a rare odontogenic tumor consisting of neoplastic epithelial and mesenchymal tissues. Recent reports have suggested that this lesion has the potential for high recurrence (18%) and greater chances of recurrent Ameloblastic fibroma transforming into Ameloblastic fibrosarcoma (45%). A 34-year-old male patient presented with pain and swelling in right mandibular posterior region. Intraorally expansion of buccal cortical plate with tenderness over swelling was present. Extraoral examination revealed facial asymmetry on right side. In view of imaging and clinical findings, provisional diagnosis of Odontogenic Keratocyst or Recurrent Ameloblastoma was considered. After obtaining informed consent and general systemic evaluation, the lesion was enucleated under general anesthesia and biopsied which confirmed the diagnosis of Ameloblastic fibroma. Ameloblastic fibroma is a mixed odontogenic tumor composed of odontogenic ectomesenchyme resembling dental papilla with epithelial strands and nests similar to the dental lamina and enamel organ, but with no dental hard tissue formation. Odontogenic tumors, Ameloblasts, Ameloblastoma, Jaw neoplasm.
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Orthokeratinised odontogenic cyst: A case series Highly accessed article p. 64
Rashmi Metgud, Anukriti, Smitha Naik, Aniruddh Tak
DOI:10.4103/jomfp.jomfp_440_21  
Orthokeratinised odontogenic cyst (OOC) is a distinct and an uncommon odontogenic cyst of jaw. It was previously considered as orthokeratinized variant of Odontogenic Keratocyst, owing to its same site, age and occurrence and origin. In 2017 WHO classified OOC under odontogenic cysts as a distinct entity, clearly separating it from Odontogenic Keratocyst (OKC). In cases of malignant transformation, the cyst shows unclear boundaries. The cases that reported to the department during the period 2019-2021 were retrieved and then reviewed by the authors. A total of 7 cases that met the histopathological criteria for OOC were included in the study. There was a clear male predilection. Age of patients ranged between 17 to 65 years. Most of cases showed Unilocular radiolucency but one case revealed Multilocular pattern. Grossly specimens ranged between 0.5 cm to 3.0cms. Histopathological findings showed all classic features of OOC. FNAC was also done and it is clearly evident that the use of Cytology can help us diagnose the cyst early and is a less invasive method. OOC is a rare type of odontogenic cyst, but while making the diagnosis one should not forget about the fact that it is less aggressive and needs to be differentiated from OKC. Majority times it has been seen that Clinico-radiographic diagnosis given of any circumscribed radiolucency around an impacted tooth is Dentigerous cyst, which is not correct. Diagnosing such lesions require thorough histopathological and radiographical correlation along with molecular analysis to reach final diagnosis.
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Calcifying odontogenic cyst associated with compound odontoma – A rare entity p. 69
BK Akshatha, GS Manjunath, N Soundarya
DOI:10.4103/jomfp.jomfp_411_22  
Calcifying odontogenic cyst (COC) or Gorlin's cyst categorized as a distinct entity by Gorlin et al. in 1962. It is a rare benign developmental cystic lesion that accounts for less than 1% of all odontogenic cysts. It can occur in association with various types of odontogenic tumors such as odontomas. COC is a hybrid lesion of the jaw presenting a manifold variety of clinical behaviors and histopathological characteristics including cystic, solid (neoplastic), and aggressive forms. COC exhibits diversity in terms of its clinical presentations, histopathologic features, and biological behavior. Normally, it presents as asymptomatic, slow developing lesion affecting the maxilla and mandible equally with strong predilection for the anterior segment. Radiographically, these lesions usually present as a unilocular, well-defined radiolucency with radiopaque structures within the lesion, either as irregular calcifications or tooth-like densities. The distinct features of COC as solid or cystic lesions presents with an ameloblastomous component admixed with varying proportions of ghost cells and spherical calcifications. Here, we report an article of calcifying odontogenic cyst associated with compound odontoma in a young patient with lesion in the anterior mandible with review of the various terminologies and classification and histopathology.
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Hypohidrotic ectodermal dysplasia: A rare entity p. 75
Himanshu Srivastava, Chintu K Singh, Sameera M. R Qureshi, Chaitra S Mastud
DOI:10.4103/jomfp.jomfp_72_21  
Hypohidrotic ectodermal dysplasia (HED) is a genetic disorder which involves a complex group of inherited conditions. This condition is characterised by the failed development of two or more ectodermal derived anatomic structure; for example, the skin, hair, nails, teeth, and sweat glands. It is a phenotypically heterogenous group of illnesses including anodontia with conical teeth, generalised spacing, reduced ability to sweat, very less hair growth, etc. The pattern of inheritance of HED varies from person to person depending on the type of ectodermal dysplasia (ED). The patterns include X-linked recessive, X-linked dominant, autosomal dominant, autosomal recessive, and spontaneous mutation. There is one such common type of ED more in female than male. It is an X-linked HED (XLHED), characterised by hypohidrosis, sparse hair, and teeth abnormalities. The other type can affect both male and female equally and may be inherited in different ways. The present case is a 21-year-old male who presented with hair, teeth, and sweat gland abnormalities.
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Bilateral peripheral facial palsy: A rare case report p. 80
Maria Luiza De Santa Mocelin, Liliane Roskamp, Natanael Henrique Ribeiro Mattos, Cintia Mussi Milani
DOI:10.4103/jomfp.jomfp_200_22  
Peripheral facial palsy (PFP) is an inflammation of the facial nerve, which paralyses the face unilaterally or bilaterally, causing pain and discomfort to the patient. PFP affects the lives of compromised individuals not only due to the loss of essential facial functions (smiling, blinking, talking) but also their emotional state. When the face is paralysed, the lost ability to animate the face can be devastating and is often associated with depression, social isolation, and reduced quality of life. Bilateral involvement is extremely rare and as it occurs in unilateral cases, a thorough clinical and laboratory evaluation must be carried out to determine the etiology of the disease, which can be idiopathic, infectious, neoplastic, traumatic, or iatrogenic. In addition to these, in times of the pandemic, coronavirus disease 2019 (COVID-19) and the vaccine against it should be considered as possible causal factors. Drug therapy and physiotherapy are indicated to recover facial movements. The aim of the present study was to report a case of bilateral peripheral facial palsy due to herpes simplex virus reactivation in a 20-year-old female patient.
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Tru-cut needle biopsy: A novel approach in the diagnosis of solid oral pathologies p. 85
Sai Lalita, Thamizhchelvan Harikrishnan, Emmanuel DS Azariah, Deepak Chandrasekaran
DOI:10.4103/jomfp.jomfp_212_22  
Nowadays, new biopsy techniques such as fine and wide needles are now employed instead of invasive biopsy techniques. Compared to open biopsy, true-cut needle biopsy has a number of advantages. It's quick and simple to do, can be done in an outpatient department, avoids incisions into previously irradiated skin, and has few risks. In order to examine malignant and benign tumours, there had been a debate in the past century on the utilisation and efficiency of tru-cut biopsy over Fine needle aspiration cytology (FNAC) and surgical biopsy in solid tumours. A non-odontogenic tumour that occurs in jaws, also classified as a fibro-osseous lesion of the jaw, is Cemento-Ossifying Fibroma. Clinically, these lesions occur as gradually growing, reaching an enormous size if not treated. In this article, a case of cemento-ossifying fibroma noticed in the maxilla with facial swelling is discussed and the diagnosis was done using a tru-cut needle biopsy.
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Atypical variant of Stafne bone defect mimicking odontogenic cyst of the jaw p. 91
T Manigandan, Rakshanaa T. V Rajalakshmi, Pavani Dornadula
DOI:10.4103/jomfp.jomfp_255_22  
Stafne first described the term “Stafne bone defects” (SBDs) in 1942. These are unilateral, asymptomatic, well-defined radiolucent lingual bony defects located at the posterior region of the mandible below the inferior alveolar canal. It is most commonly seen in the posterior region, whereas it is relatively rare in the anterior region. This anterior variant of SBD is often misdiagnosed as any other odontogenic cyst, and hence advanced imaging techniques should be followed to identify it at the earliest. Due to the lower prevalence of this entity, only a handful of cases have been documented for the anterior variant of SBD. In this paper, we have thus documented the rare variant of SBD in the anterior region.
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Lower lip cysticercosis: An unusual case report p. 95
Mala Sagar, Shalini Rawat, Anurag Singh, Anuragini
DOI:10.4103/jomfp.jomfp_329_22  
Cysticercosis is a disease that occurs when humans are infected with Taenia solium larvae. The parasitic illness cysticercosis, which is common in impoverished nations, hardly ever affects the mouth. Oral cysticercosis is rare that might be challenging to diagnose clinically. This rare case report documents an oral cysticercosis case in a 38-year-old male patient who had an asymptomatic lesion in the buccal mucosa of his lower lip. A T. solium larva was identified on histopathological examination in a cystic cavity after an excisional biopsy.
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Adult-onset Langerhans cell histiocytosis – A Trojan horse of oral cavity: A case report with rare clinical presentation p. 98
CJ Sanjay, Karthikeya Patil, Usha Hegde, Romali Panda
DOI:10.4103/jomfp.jomfp_131_22  
Langerhans cell histiocytosis (LCH), earlier recognised as histiocytosis X, is a rare haematological illness involving infants and young children. LCH is caused by unrestrained stimulation and proliferation of usual antigen presenting cells, Langerhans cells (LCs) and the disease demonstrates extensive clinical and radiographic features involving multiple sites. Since the incidence is relatively low limited data is available regarding the epidemiology of LCH, with approximation of 2–5 cases per million populations per year. LCH has male predilection with jaws involved in 10–20% cases and only 1% of the cases affecting maxilla, masquerading as periodontal or periapical pathology. We report a case of 48-year-old female with LCH involving posterior maxilla. This is a unique presentation corresponding to age, gender, location and severity. Dental clinicians should be aware of this and consider it to be a part of their differential diagnosis pertaining to unresolved periodontal pathology as it mimics clinically and radiographically.
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Rapidly expanding chondromyxoid fibroma of the mandible: A case report of rare entity p. 104
Gargi Jadaun, Himanshu Gupta, Sayeeda Kharodia, Viral Gadhiya
DOI:10.4103/jomfp.jomfp_351_21  
Chondromyxoid fibroma is an unusual, benign bone tumour that is usually sited in the metaphyseal region of the long bones. It accounts for less than 1% of all bone tumours. It manifests predominantly in males in their second and third decades of life. Rarely, it occurs in the bones of the craniofacial skeleton. For small, lesions enucleation and curettage and for larger lesions, resection followed by are the treatment modalities available. Here, we present a case of chondromyxoid fibroma with respect to the right side of the mandible which was managed by surgical resection followed by reconstruction under general anaesthesia. Resection followed by reconstruction provides satisfactory outcomes, especially in cases with large lesions. Chondromyxoid fibroma is an asymptomatic, benign, slow-growing lesion but can rapidly expand and involve the greater area of bone. Thus, its detection at an early stage and treatment at the proper time can lead to less morbidity associated with the lesion and improved quality of life of the patient.
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