Ameloblastoma With the Hybrid Desmoplastic and Plexiform Pattern: A Case Report.

Ameloblastoma is an epithelial odontogenic tumor with a benign nature and demonstrates local aggressiveness. It frequently occurs between the third and fifth decades of life, showing significant gender predilection. While typically displaying a benign growth pattern, it tends to invade and sporadically metastasize locally. Ameloblastoma is predominantly found in the posterior regions. Periodic recur commonly follows insufficient treatment. Hence, conducting thorough identification of tumors and management is crucial to prevent relapse. Complications and improved prognosis are associated with meticulous surgical techniques, regular follow-up care, and early detection of recurrence. This study presented a report of a 19-year-old male with swelling in the left lower jaw, detailing its area of complaint, radiographic findings, histopathologic characteristics, and different treatment approaches. The uniqueness of the case is the hybrid histopathology of ameloblastoma composed of plexiform and desmoplastic variants.

Desmoplastic histological subtype of ameloblastoma in 16 dogs.

Ameloblastoma in dogs most often has a highly conserved acanthomatous cell morphology and is recognized as canine acanthomatous ameloblastoma (CAA) while conventional ameloblastoma (CA) makes up a smaller yet morphologically diverse group of epithelial odontogenic tumors. In humans, a rare desmoplastic histological subtype has distinctive clinical, radiological, and microscopic features. Desmoplastic ameloblastoma (DA) has not previously been described in dogs, although it has been rarely referenced in the veterinary literature. This is the first thorough description of a case series of DA in dogs and describes clinical presentation, diagnostic imaging findings, histopathological features for diagnosis, and treatment outcome. Clinically, DA most often presents as a mass or swelling in the rostral mandible or maxilla of middle age to older dogs. On diagnostic imaging, the lesion has a radiolucent or mixed pattern with well-defined borders and variable loculation. As a solid, fibrous tumor with obscured odontogenic epithelium, DA is challenging to diagnose histologically and can mimic several other oral tumors, both benign and malignant. As an ameloblastoma, the biological behavior of DA is locally destructive yet benign and prognosis is favorable following surgical excision.

Collision Tumour Comprising Desmoplastic Ameloblastoma and Squamous Odontogenic Tumour in the Anterior Maxilla: A Case Report.

Collision tumours, characterized by the simultaneous occurrence of two distinct neoplasms within the same anatomical site, are exceedingly rare in oral pathology. This case report presents an uncommon collision tumour involving desmoplastic ameloblastoma and squamous odontogenic tumour in the anterior maxilla of a 52-year-old male from the Indian population. Desmoplastic ameloblastoma is a variant of ameloblastoma known for its unique histopathological features, while squamous odontogenic tumour is a benign epithelial odontogenic tumour with distinctive clinical behaviour. The rarity of this occurrence emphasizes the need for accurate diagnosis and effective treatment strategies. This report discusses the clinical presentation, radiographic findings, and histopathological characteristics of this collision tumour. Through the presentation of this case, we aim to contribute to the understanding of these rare entities and their management considerations.

Desmoplastic Ameloblastoma-A Case Report.

Desmoplastic ameloblastoma (DA) is a rare variant of conventional ameloblastoma. It accounts for only 4%-13% of all ameloblastomas. DA was included in the World Health Organization Classification of Head and Neck Tumors (WHO-2005) as a variant of ameloblastoma with specific clinical, imaging, and histological features. The desmoplastic variant of ameloblastoma usually appears in the anterior and premolar regions as a mixed radiolucent and radiopaque lesion, sometimes resembling a benign fibro-osseous lesion.Ameloblastoma is a locally aggressive tumor that may cause recurrence and in rare cases, malignant transformation with repeated postsurgical recurrences. In this paper, we present a case of a 28-year-old female with swelling in the left upper jaw, a biopsy of which turned out to be DA.

Desmoplastic ameloblastoma with mucous cell differentiation: A Rare case report.

Ameloblastomas are tumors arising from the odontogenic epithelium. Ninety cases of desmoplastic ameloblastoma have been reported so far in the literature, out of which only five cases with mucous cell differentiation have been reported. We are presenting a case of 24-year-old female having a chief complaint of a painless swelling on the left side of the face for 7 months. After radiological, histopathological findings lesion was diagnosed with unicystic ameloblastoma which was treated by segmental resection. The purpose of this article is to present a case of desmoplastic ameloblastoma that has occurred in an unusual site and has unique histopathological features.

Unicystic desmoplastic osteo-ameloblastoma of posterior mandible: Report of rarest of the rare case and review of literature.

Desmoplastic ameloblastoma (DA) is an epithelial odontogenic tumor and a rare variant of ameloblastoma. It was first described by Eversole in 1984. In the World Health Organization (WHO) classification of odontogenic tumors (2005), DA has been considered as a distinct entity from conventional/multicystic ameloblastoma. DA differs strikingly in its clinical, radiological, and histopathological presentation when compared to other variants of ameloblastoma. We report here an extremely rare "Hybrid DA" in a 50-year-old female patient with painless hard swelling involving right posterior mandible with detailed clinical history, an unusual radiographic and histopathological presentation. Histopathology revealed odontogenic epithelium in the form of follicles, ameloblastoma with cystic degeneration, and squamous metaplasia at places and elsewhere there were odosntogenic islands compressed by dense fibrocellular stroma suggestive of desmoplasia along with osseous tissue formation. Also review of the literature and possible explanation of etiopathogenesis of cystic change and osseous tissue formation in DA are discussed.

Desmoplastic ameloblastoma of anterior maxilla - A case report.

The recently recognized desmoplastic ameloblastoma is considered a rare variant of central ameloblastoma. It has been included in the World Health Organization's histopathological typing of odontogenic tumors, similar to benign, locally invasive tumors with low recurrence rate and peculiar histological features, characterized by epithelial changes due to the pressure of stroma on epithelial tissue. The aim of this paper is to report a unique case of desmoplastic ameloblastoma in the mandible of a 21-year-old male with a painless swelling in the anterior region of the maxilla. To our knowledge, only a few cases of adult patients affected by desmoplastic ameloblastoma have been published.

Retrospective analysis of Desmoplastic Ameloblastoma: Clinical review / Análisis retrospectivo del ameloblastoma desmoplásico: Revisión clínica

Background: Desmoplastic Ameloblastoma (DA) is a rare, true neoplasm of jaws with reported incidence of4-13% among other variants of Ameloblastoma, however this appears distinct than the classic Ameloblastoma inanatomical distribution and clinical presentation. This is often mistaken as a fibro-osseous lesion because of itssimilar radiological appearance.Material and Methods: To describe the clinical, radiographic and histopathological characteristics through a se-ries of new cases of histologically proven DA including a case of an exceptionally large, recurrent lesion alongwith retrospective analysis of cases from literature available for an improved understanding of the behaviour andprognosis of DA. A total of 50 cases were analysed for the anatomical distribution, radiographic presentation andmanagement. Out of the 50 cases, 47 cases were from the English literature reported from 2011 to 2019 and 3 werenew cases.Results: DA showed a slight male predilection (male: female=1.17:1) with a predominance in the fourth and fifthdecade of life. Mandibular involvement (52%) was more commonly seen with a marked tendency for the anteriorregion. Radiographically, most of the lesions presented mixed radiopacity with radiolucency(80%) and root dis-placement was observed in only 70.27 % cases. Recurrence rate of 26 .47 % was observed. Cases treated withresection resulted in lesser recurrence as compared to those treated with enucleation and curettage. Conclusions: DA is distinguished by a peculiar display of clinicalopathological parameters. DA has tendency of lo-cal disposition and propensity of recurrence, which thus necessitates its aggressive management. It is not possible toconclude or report on the aggressive/recurrent nature and appropriate treatment modality for DA due to inadequatefollow-up results.(AU)

Desmoplastic ameloblastoma: A case report.

Desmoplastic ameloblastoma (DA) is a rare variant of ameloblastoma, accounting for approximately 4 to 13% of ameloblastomas. It is uncommon, aggressive in nature, and there are high chances of misdiagnosis. Clinical and radiographical features are similar to fibro-osseous lesions of jaw. We reported a case of 35-year-old male patient of DA.

Desmoplastic ameloblastoma of the jaw: CT and MR imaging findings.

OBJECTIVES: Desmoplastic ameloblastoma (DA) is one of the rare pathological variants of ameloblastoma. The purpose of this study is to investigate CT and MR imaging findings of DA. METHODS: We retrospectively evaluated six DA cases that were gathered from four different hospitals and confirmed histopathologically with resected specimens. Two radiologists who specialized in head and neck imaging retrospectively interpreted and reviewed pretreatment CT and MR imaging findings. RESULTS: On CT, all DAs presented with well-defined border and low internal attenuation. Regarding locularity, five cases showed honeycomb-like lesions and one showed a unilocular lesion. Bone expansions were seen in all patients and located on the labial side in four cases. On MR imaging, all DAs showed well-defined borders and solid moderately low signal intensities with small cystic high signal intensities on T2-weighted images and intermediate signal intensity on T1-weighted images. Five DAs showed linear low signal intensity on T1- and T2-weighted images. Gd-DTPA-enhanced MR imaging was performed in three cases, and all cases showed moderate enhancement. Dynamic-enhanced MR imaging was performed in two of them, and persistent enhancement was detected. CONCLUSION: Our findings strongly suggest that MR imaging should also be consulted, particularly for honeycomb-like lesions with well-defined margins, bone expansion and anterior location on radiographs or CT. Additionally, findings by MR imaging such as solid low-signal-intensity, hyperintense cystic foci and persistent enhancement pattern will suggest the diagnosis of DA.

Desmoplastic ameloblastoma: a systematic review of the cases reported in the literature.

The aim of this study was to integrate the available data published on desmoplastic ameloblastoma (DA) into a comprehensive analysis of its clinical/radiographic features. As it is not clear whether the hybrid ameloblastoma has a unique biological behaviour, the clinical/radiographic features of conventional DA were compared with those of the hybrid variant. An electronic search was undertaken in May 2019. Eligibility criteria included publications with sufficient clinical/radiographic/histological information to confirm the diagnosis. A total of 128 publications reporting 285 DAs were included (246 central non-hybrid, 33 central hybrid, 6 peripheral). There was a statistically significant difference between non-hybrid and hybrid cases concerning lesion location, radiographic limits, and size. In contrast to non-hybrid tumours, which showed a similar distribution in the mandible and maxilla, hybrid tumours showed a high predominance of mandible location. Non-hybrid lesions more often showed ill-defined radiographic margins, and were smaller tumours than hybrid DAs. Enucleation with an additional therapy (either curettage or peripheral osteotomy) or resection led to a lower recurrence risk in comparison to curettage or enucleation without additional therapy. The location of the lesion and type of treatment are related to tumour recurrence. There are important differences in location and radiographic limits between hybrid and non-hybrid DAs, which support their classification as distinct lesions.

A mixed radiopaque - radiolucent lesion in the anterior mandible associated with multiple impacted teeth: A radiodiagnostic challenge?

Desmoplastic ameloblastoma (DA) exhibits important differences in gender, anatomic distribution, radiographic findings, and histologic appearance compared to other types of ameloblastoma. Radiologically, DA is seen either as ill-defined mass containing osteolytic and sclerotic areas or as multifocal radiodense flecks within radiolucent background resembling a honeycomb. The radiographic differential diagnosis includes fibro-osseous lesions such as cemento-ossifying fibroma, fibrous dysplasia, calcifying odontogenic cyst, and chronic sclerosing osteomyelitis. Thus, DA should primarily be included in the differential diagnosis of a mixed radiopaque-radiolucent lesion with diffuse borders in the anterior premolar region of the jaws. This report adds to the literature of mixed radiolucent-radiopaque lesions which may not always be histopathologically diagnosed as a fibro-osseous lesion but could turn out to be a DA. This report also benefits the dental community by cautioning them to be aware of DA that can be associated with multiple unerupted teeth which is quite a rare finding.

Hybrid Ameloblastoma of Anterior Maxilla: A Rare and Puzzling Pathologic entity - Case Report with Systematic Review.

Hybrid ameloblastoma has a variable clinical, radiological, and histopathological presentation. They contain two or more different histologic types and their biologic comportment is still arguable. We herein present a case of a hybrid variant of desmoplastic ameloblastoma which is the first of its kind to have ever been reported due to its unusual location in the maxillary anterior region, along with systematic review of clinicopathologic features of reported cases immunohistochemical markers may act as an adjunct in the accurate diagnosis of these lesions.

Malignant Transformation of a Desmoplastic Ameloblastoma to Squamous Cell Carcinoma: A Case Report.

Ameloblastomas are the most common odontogenic tumors, excluding odontomas. Several morphologic variants have been described including follicular, plexiform, acanthomatous, granular cell, basaloid and desmoplastic. Desmoplastic ameloblastoma differs from other conventional ameloblastomas microscopically, clinically, and radiographically. Ameloblastic carcinoma, the malignant counterpart of ameloblastoma is characterized by cytologic features of malignancy combined within the overall histologic features of conventional ameloblastoma. Malignant transformation of ameloblastoma to squamous cell carcinoma is a controversial subject. Here we report a case of a desmoplastic ameloblastoma with malignant transformation to squamous cell carcinoma in a 49 year old African American man. The patient underwent tumor resection and radiation therapy with no evidence of disease recurrence or progression 16 months post operatively. To our knowledge malignant transformation of a desmoplastic ameloblastoma to squamous cell carcinoma has not so far been reported. This observation may lend some support to the argument that desmoplastic ameloblastoma is phenotypically and biologically distinct entity.

Clinicopathological characteristics of desmoplastic ameloblastoma: A systematic review.

The aim of the present review was to systematically present the clinicopathological data of desmoplastic ameloblastoma (DA) from articles published in the literature. A comprehensive search of the databases (PubMed, Medline, SCOPUS, Web of Science, and Google Scholar) for published articles on DA was conducted. A total of 238 cases were identified and analyzed from 76 published papers. DA showed a slight male predilection (male: female=1.07:1) with a predominance in the fourth and fifth decades of life. Mandibular involvement (52.55%) was most commonly seen with a marked tendency for the anterior region (mandible: 40.9%, maxilla: 48.07%). The size of the lesion ranged from .5 cm to 20.4 cm, with the majority of cases measuring more than 3 cm in size (53.84%). Radiologically, most of the lesions presented mixed radiolucency and radiopacity (62%), and root resorption was observed in only seven cases. The majority of the lesions showed ill-defined margins upon radiographic examination (65.78%). Most of the cases were treated with resection (78.57%), and five of the 10 recurrent cases were treated by enucleation/curettage. DA is characterized by the unique presentation of clinicopathological parameters. It is not possible to comment on its aggressive/recurrent nature and best treatment modality due to inadequate follow-up data.

A case report on desmoplastic ameloblastoma of anterior mandible.

BACKGROUND: Desmoplastic ameloblastoma (DA) is a rare variant that accounts for approximately 4-13% of ameloblastoma, displaying significant differences in anatomical site, imaging, and histologic appearance. It has been included in WHO classification of head and neck tumor (WHO-2005) as a variant of ameloblastoma. The tumor resembles benign fibro-osseous lesion for being frequently occurring in the anterior region of jaws as a mixed radiopaque-radiolucent lesion. CASE PRESENTATION: We present a case of DA in a 43-year-old female with a painless swelling in the anterior region of mandible. No fluid was evident on fine needle aspiration. A mixed lesion with multilocular appearance was evident on both panoramic radiographs as well as computed tomography scan. An incisional biopsy confirmed it to be a case of desmoplastic ameloblastoma. Segmental mandibulectomy was performed from teeth 35 to 44. The patient is on routine follow-up and is currently free of ailment. CONCLUSIONS: The present case deserves emphasis because of its unfamiliar appearance, potentially aggressive nature and deceptive radiologic appearance maximizing the chances of misdiagnosis. So, the clinician should be alert enough to include desmoplastic ameloblastoma in differential diagnosis of any lesion/growth with mixed radiolucent-radiopaque appearance having ill-defined borders and occurring in anterior maxilla or mandible.

Ameloblastoma desmoplásico: revisão de literatura / Desmoplastic ameloblastoma: review of the literature

O ameloblastoma desmoplásico é uma variante dos ameloblastomas e caracteriza-se por estroma densamente colagenizado e permeado por pequenas ilhas e cordões de epitélio tumoral odontogênico com pouca tendência para formar estruturas císticas. Clinicamente apresenta-se como um aumento volumétrico facial indolor localizado predominantemente na região anterior da mandíbula; possui pequena predileção pelo sexo masculino e baixa incidência entre os tumores odontogênicos. Radiograficamente é descrito como uma lesão radiolúcida de limites pouco nítidos, semelhante a bolhas de sabão e que pode mimetizar lesões fibro-ósseas. Reabsorção radicular e neoformação óssea podem estar presentes. A tomografia computadorizada e a ressonância magnética nuclear são exames de imagem úteis para o planejamento do tratamento, principalmente devido às margens mal definidas da neoplasia. O tratamento é controverso, mas a maioria dos autores corrobora a necessidade de excisão com adequada margem de tecidos não envolvidos devido ao alto índice de recidivas. Assim, esse trabalho teve como propósito revisar a literatura pertinente ressaltando as especificidades dessa lesão rara. De acordo com a literatura consultada pudemos concluir que as particularidades dessa variedade de ameloblastoma devem ser conhecidas e que, apesar da controvérsia, o tratamento mais seguro ainda é a ressecção com margens de aproximademente 1 cm...

The desmoplastic ameloblastoma is a variant of ameloblastomas and is characterized by densely collagenized stroma and permeated by small islands and strands of odontogenic tumor epithelium with little tendency to form cystic structures. Clinically presents as a painless facial volumetric increase localized predominantly in the anterior mandible; there is a little preference for male and low incidence among odontogenic tumors. Is described radiographically as a radiolucent lesion with ill-defined borders, similar to soap bubbles and can mimic fibro-osseous lesions. Root resorption and bone formation may be present. Computed tomography and magnetic resonance are useful exams for treatment planning, especially due to ill-defined margins of the neoplasm. Treatment is controversial, but most authors corroborates the need for excision with adequate margin of uninvolved tissue due to the high rate of recurrence. Thus, this work aimed to review the literature emphasizing the specifics of this rare lesion. According to the literature we concluded that the characteristics of this variety of ameloblastoma should be known and that despite the controversy, the safest treatment still is the resection with margins of approximately 1 cm...

Peripheral desmoplastic ameloblastoma in adolescent age: clinico-pathological and immunohistochemical analisys of a case.

The Extraosseous or Peripheral Ameloblastoma (PA) is a rare and benign odontogenic tumour, representing 1% to 5% of all ameloblastomas. It is usually localized in the soft oral tissues, without deep bone involvement. Its biological behaviour is specific, and several authors define PA as a non-infiltrating hamartomatous lesion. Indeed, recurrences rarely occur and progression in malignant tumors appears to be rare. The PA originates from the tooth-forming apparatus and it consists of proliferating odontogenic epithelium, exhibiting the same histological cell types and patterns of the intraosseous counterpart or infiltrating ameloblastoma. The peripheral desmoplastic ameloblastoma (PDA) can be classified as a newly recognized and very rare histological variant. To our knowledge, only a few cases of adult patients affected by PDA have been published. The aim of this paper is to report a case of PDA affecting an adolescent patient. The clinical-pathological and immunohistological features are discussed in order to improve knowledge regarding a correct diagnosis and to differentiate PDA lesions from similar diseases.

A case of desmoplastic ameloblastoma occupying maxillary sinus.

We report a rare case of desmoplastic ameloblastoma lesion that filled the entire maxillary sinus. The patient visited our hospital with a chief complaint of swelling around the upper left premolars. A panoramic X-ray captured an image of a mixture of ill-defined radiolucency and radiopacity from the swollen area to the maxillary sinus. Computed tomography (CT) and Magnetic resononce imaging (MRI) showed that the lesion occupied almost the entire left maxillary sinus and had entered the nasal cavity. A pathologic diagnosis of ameloblastoma was made after biopsy, and the tumor was removed and the marginal bone curetted under general anesthesia. A CT scan at 4 months postoperatively indicated the presence of residual and recurrent tumor in the area of the upper left lateral incisor, and removal and curettage were performed again. Recurrence may be detected relatively easily based on radiographic characteristics, and therefore follow-up with an X-ray examination such as a CT scan is important.

Desmoplastic ameloblastoma: a case report.

Desmoplastic ameloblastoma is a rare variant of ameloblastoma. Up until now, less than 150 patients have been reported in the literature. We report a case of desmoplastic ameloblastoma in a 45-year-old female with a painless swelling in the left anterior maxillary region. Fine needle aspiration yielded no fluid. Periapical and panoramic radiographs as well as computer tomography scan showed a mixed lesion with multilocular appearance. The present case deserves special importance be-cause of its unfamiliar appearance, potentially aggressive nature and high chances of misdiagnosis. Moreover, the radio-graphic features of this lesion rarely point towards ameloblastoma. A partial maxillectomy for tumor resection was per-formed and the involved teeth were removed. This report is an attempt to help the dental community in developing familiarity with the clinical presentation and at the same time advocating to develop a high index of suspicion in recognizing such cases.