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Patterns of expression of intermediate filaments and S-100 protein in desmoplastic ameloblastoma

Siar CH, Ng KH

J Nihon Univ Sch Dent, 1993 Jun;35(2):104-8.
PMID: 7692015

Seventeen cases of desmoplastic ameloblastoma were examined immunohistochemically. Immunoperoxidase techniques were applied for detection of keratin, desmin, vimentin and S-100 protein expression in these tumors. The tumor epithelium of desmoplastic ameloblastoma exhibited weak, focal, inconstant keratin staining, weak, variable expression of S-100 protein, desmin immunoreactivity of mild to moderate intensity and vimentin non-reactivity. The pertinent literature on the immunohistochemistry of ameloblastomas is briefly reviewed.

Matched MeSH terms: Ameloblastoma/immunology; Ameloblastoma/pathology; Ameloblastoma/chemistry*
Fulltext Management of large mandibular ameloblastoma - a case report and literature reviews

Kahairi, A., Ahmad, R.L., Wan Islah, L., Norra, H.

Archives of Orofacial Sciences, 2008;3(2):52-55.
MyJurnal

Ameloblastoma is a slow growing benign tumour of the jaw and patients usually present late after the tumour achieved considerable size to cause facial disfigurement. Diagnosis mainly from tissue biopsy and characteristic findings on plain X-rays does assist in differentiating between types of ameloblastoma. The challenges in the management of this tumour are to provide complete excision as recurrence may occur in incomplete removal and also to reconstruct the bony defect in order to give reasonable cosmetic and functional outcome to the patient.

Matched MeSH terms: Ameloblastoma
Desmoplastic ameloblastoma in Indians: report of five cases and review of literature

Sivapathasundharam B, Einstein A, Syed RI

Indian J Dent Res, 2007 10 17;18(4):218-21.
PMID: 17938502

In the recent World Health Organization classification of odontogenic tumours, desmoplastic ameloblastoma has been characterized as a variant of ameloblastoma, with specific clinical, radiographical, and histological features. Till date, 145 cases have been reported in Japanese, Chinese, Malaysian, Western, and African populations, with very few cases described in Indians. Here, we report five cases in the Indian population. The male to female ratio was 3:2. The mean age at diagnosis was 33.2 years. Four of the tumours were located in the maxilla, in the anterior premolar region. The lone mandibular tumour was located anteriorly, crossing the midline. Three of the tumours had a mixed radiologic appearance with poorly defined borders. Unilocular change was seen in one of them. Two tumours presented as unilocular radiolucencies with specks of radiopacities and well-circumscribed borders. Histologically, irregular odontogenic islands, with a stretched-out 'kite-tail' appearance, were seen in a dense desmoplastic stroma. The peripheral layer of the epithelial islands was made up of flattened cells and the inner core was made up of spindle-shaped and, in some instances, squamous-shaped cells. In two cases, odontogenic epithelium in the form of follicles, typical of solid/multicystic ameloblastoma, was seen and these were typed as 'hybrid' variants. All the cases were treated by resection.

Matched MeSH terms: Ameloblastoma/diagnosis*; Ameloblastoma/pathology
Fulltext Unicystic ameloblastoma: a new entity?

Zain R

Med J Malaysia, 1986 Dec;41(4):370-3.
PMID: 3670162

This article consists of two selected case reports of a recently named odontogenic tumour, unicystic ameloblastoma. The clinical and radiographic findings of the two cases mimic that of odontogenic cysts but not dentigerous cysts as in most reported, cases. Histologically, either a normal or ameloblastomatous cyst lining is evident. Other features of ameloblastoma are present within the cyst wall or as luminal nodules within the cystic space. A review of the literature indicates that this is a non-aggressive tumour with a low recurrence rate.

Matched MeSH terms: Ameloblastoma/pathology*; Ameloblastoma/radiography
An immunohistochemical study of two cases of either peripheral odontogenic fibroma (WHO type) or peripheral ameloblastoma

Chong Huat Siar, Kok Han Ng

J Nihon Univ Sch Dent, 1996 Mar;38(1):52-6.
PMID: 8648412

Two cases of either peripheral odontogenic fibroma (POF) (WHO type) or peripheral ameloblastoma are reported. Their immunohistochemical characteristics were investigated in an attempt to clarify their histogenesis. The results showed that the epithelial component of this neoplasm tended to retain its distinct odontogenic character and expressed a keratin profile different from that of the overlying oral epithelium from which both cases most probably originated. The connective tissue element of these tumors was vimentin-positive and S-100 protein negative, confirming their mesodermal nature but precluding the possibility of ectomesenchymal derivation. No reactivity for desmin was noted.

Matched MeSH terms: Ameloblastoma/pathology; Ameloblastoma/chemistry
Fulltext Ameloblastomas--a clinicopathologic study of 133 cases in peninsular Malaysia

Ramanathan K, Chelvanayagam PI, Ng KH, Ramanathan J

Med J Malaysia, 1982 Mar;37(1):18-24.
PMID: 7121342

Ameloblastomas formed 1.1 percent of all oral pathology cases reported. The race, sex and age group distribution of 133 cases are shown. The peak age incidence (70.6 percent) was between 11-40 years. The mandible was involved 9 times more commonly than the maxilla. The anatomical sites of distribution, clinical and radiological features, histological variants and their correlation are discussed. Twenty two patients (15 percent) had ameloblastomas associated with a dentigerous cyst and/or unerupted teeth. Ameloblastomas with the above clinical features represented a much less aggressive form of neoplasm. The authors could not correlate histological variants of ameloblastoma with recurrence rates. The various treatment methods and the respective recurrence rates are outlined. Radiotherapy and marsupialization as treatment of ameloblastoma are not recommended. The indications for enucleation curettage, resection en bloc, segmental resection and hemimandibulectomy
are emphasized. Ameloblastomas involving the maxilla should be treated by complete removal en bloc with a margin of normal tissue. Since ameloblastoma has the capacity to recur after several years of apparent cure patients who have been treated for ameloblastoma must be followed up periodically during their life time. So far no case of ameloblastoma in this study has shown evidence
ofmetastasis.

Matched MeSH terms: Ameloblastoma/pathology*; Ameloblastoma/therapy
Unusual granular cell odontogenic tumor. Report of two undescribed cases with features of granular cell ameloblastoma and plexiform granular cell odontogenic tumor

Siar CH, Ng KH

J Nihon Univ Sch Dent, 1993 Jun;35(2):134-8.
PMID: 7692017

Granular cell ameloblastoma (GCA) is a well recognized variant of follicular ameloblastoma with extensive granular cell change. In contrast, plexiform granular cell odontogenic tumor (PGCOT) is a rare and recently described lesion characterized histologically by a monophasic plexiform pattern of granular cells. In this paper, two cases of an unusual granular cell odontogenic tumor exhibiting combined features of these two entities are described along with their immunohistochemical characteristics. The granular cells of both the GCA and PGCOT areas showed similar patterns of expression for keratin and S-100, which differed from those of typical ameloblastoma. No reactivity for desmin or vimentin was noted. The histomorphologic and immunohistochemical features of these hybrid tumors suggest that the granular cells present have a common origin, most probably the odontogenic epithelium.

Matched MeSH terms: Ameloblastoma/pathology*; Ameloblastoma/chemistry
Ameloblastoma of the jaws: a retrospective analysis of 340 cases in a Malaysian population

Siar CH, Lau SH, Ng KH

J Oral Maxillofac Surg, 2012 Mar;70(3):608-15.
PMID: 21723654 DOI: 10.1016/j.joms.2011.02.039

Ameloblastoma of the human jaw is an uncommon but clinically significant odontogenic epithelial neoplasm. The aim was to analyze the clinicopathologic characteristics of ameloblastoma in a Malaysian population.

Matched MeSH terms: Ameloblastoma/epidemiology*; Ameloblastoma/pathology
RANK, RANKL, and OPG in recurrent solid/multicystic ameloblastoma: their distribution patterns and biologic significance

Siar CH, Tsujigiwa H, Ishak I, Hussin NM, Nagatsuka H, Ng KH

Oral Surg Oral Med Oral Pathol Oral Radiol, 2015 Jan;119(1):83-91.
PMID: 25446507 DOI: 10.1016/j.oooo.2014.09.017

To determine the distribution patterns of bone resorption regulators, receptor activator of nuclear factor κ-B (RANK), RANK ligand (RANKL), and osteoprotegerin (OPG) in recurrent ameloblastoma (RAs) and to clarify their impact on the biologic behavior of these neoplasms.

Matched MeSH terms: Ameloblastoma/pathology*; Ameloblastoma/surgery
Premolar Cystic Ameloblastoma in a Child

Pathak S, Sonalika WG, Hs V, Tegginammani AS

J Coll Physicians Surg Pak, 2017 Jan;27(1):47-48.
PMID: 28292369 DOI: 2521

Mandibular swellings may occur as a result of many benign lesions of odontogenic or non-odontogenic origin. Ameloblastomas are benign tumours of odontogenic origin, whose importance lies in its potential to grow into enormous size with resulting bone deformity, it is a slow-growing, persistent, and locally aggressive neoplasm. The unicystic ameloblastoma (UA) represents an ameloblastoma variant, presenting as a cyst clinically and radiographically, but showing typical ameloblastomatous epithelium lining histologically. It commonly occurs in second and third decades of life and is rare in children under 12 years of age, and better response to conservative treatment. It shares many clinical and radiographic features with odontogenic cysts/tumours and/or periapical disease of endodontic origin. Reported here is an unusual case of unicystic ameloblastoma involving the crown of an unerupted mandibular first premolar in a 9-year boy in an uncommon location, which was misdiagnosed as periapical lesion of inflammatory origin clinically, and as a dentigerous cyst radiographically. This highlights the importance to routinely submit the removed surgical specimen for histopathological examination.

Matched MeSH terms: Ameloblastoma/diagnosis; Ameloblastoma/pathology*; Ameloblastoma/surgery*
Fulltext Radiological features of different histopathological variants of ameloblastomas

Chen, Y.N., Nambiar, P.

Malaysian Dental Journal, 2008;29(1):14-19.
MyJurnal

The aim of present study was to determine if the radiological features noticed on dental panoramic radiographs can ascertain the different variants of ameloblastomas. Methods: A total number of 177 cases of ameloblastoma diagnosed in 5 centers in Malaysia were reviewed. The clinical records of these cases were analyzed with respect to age, sex, ethnicity, diagnosis of the lesions, anatomic site, size, side, year of diagnosis, status of the lesion, and characteristics of the lesions. Detailed radiographic features such as septation, calcification, effect on adjacent structures, periosteal reactions, demarcation and cortication of the border of the lesions were recorded. Results: The unicystic, plexiform and acanthomatous ameloblastomas tend to have unilocular radiolucencies as opposed to the follicular ameloblastoma, which demonstrated commonly multilocular radiolucency with soap-bubble appearance. The well-demarcated borders with thin condensed sclerotic border of the lesions appeared to be more significant in unicystic ameloblastoms. Moderately and poorly demarcated lesions are commonly found in follicular and acanthomatous ameloblastomas. Multiplanar pattern of root resorptions, protrusion of roots into lesions and cortical expansions with intact visible margins are commonly seen in ameloblastomas. Conclusion: The various histopathological patterns are not closely related to any specific radiological appearances in the jaws and there seems to be no direct or obvious correlation between the histopathological patterns of the tumours and the radiological appearance of ameloblastomas.

Matched MeSH terms: Ameloblastoma
Fulltext Fibula Osteocutaneous Flap for Mandible Reconstruction after Ameloblastoma Resection: Amending Technique to Reduce Ischaemic Time

Chee, E.K., Sara Ahmad, T., Ng, E.S.

Malays Orthop J, 2007;1(1):39-41.
MyJurnal

A 27-year-old patient with ameloblastoma of the mandible
underwent anterior mandibulectomy and reconstruction with
left fibula osteocutaneous flap. The bone was shaped at the
lower limb before cutting the pedicle. Vascularised free fibula flap provides a good alternative to other bone grafts in mandible reconstruction as it is relatively easy to perform, carries a low complication rate, and mandibular shaping with intact pedicle cuts down on ischaemic as well as total operative time.

Matched MeSH terms: Ameloblastoma
Fulltext A Comparative Histopathological Study Of Epithelial Linings Of Odontogenic Cysts And Unicystic Ameloblastomas

Sudiono, J., Zain, R.B.

Ann Dent, 1998;5(1):-.
MyJurnal

The epithelial cystic linings and adjacent connective tissues of 61 cases of odontogenic cysts (radicular cysts[RC], dentigerous cysts[DC] and odontogenic keratocysts[OKC]) and unicystic ameloblastomas(UA) were described and compared histopathologically. The type of epithelium in relation to the presence of rete processes and the distribution of chronic inflammatory cells were analyzed statistically. Significant associations between the presence of rete processes in the non-keratinized epithelial linings and inflammation in the subjacent connective tissues of RC and DC were found in this study. There was also a statistically significant association between the presence of rete Processes and nonkeratinized epithelial linings in OKC. The results also showed that in inflamed OKC, the cystic lining epithelium exhibited hyperplasia indistinguishable from lining epithelium of RC and DC. This study further showed that ameloblastomatous-like epithelial cystic linings were present in inflammed odontogenic cysts. All except for one case of unicystic ameloblastomas in this study showed ameloblastomatous epithelial cystic linings. It is recommended that the lining epithelium of RC and DC be examined carefully in order to rule out OKC. Similarly, ameloblastomatous-like lining epithelium arising from chronic inflammation in RC and DC should be differentiated from true ameloblastomatous cystic lining. Such careful examinations are diagnostically important in view of the similarities of epithelial cystic linings of inflamed OKC with DC and RC aggressive behavior ofOKC and UA.

Matched MeSH terms: Ameloblastoma
Fulltext Ameloblastic carcinoma: A case with cervical node and pulmonary metastases

Khoo SP, Ong ST

Ann Dent, 1998;5(1):49-52.
MyJurnal

Odontogenic carcinomas of the jaws are subclassified into malignant ameloblastoma, ameloblastic carcinoma and primary intraosseous carcinoma arising from within the bone. These may arise from residual islands of epithelium derived from dental lamina or epithelial lining of dental cysts. Ameloblastic carcinoma is extremely rare. An aggressive case of ameloblastic carcinoma occumng in a 59-year-old Malay man is presented. Wide excision of the primary lesion with radical neck dissection was carried out. He developed lung metastasis 4 months post-operatively. Despite chemotherapy upon discovery of lung metastasis, he expired 7 months following the initial diagnosis.

Matched MeSH terms: Ameloblastoma
Fulltext Assessment of fragile histidine triad expression in ameloblastoma, odontogenic keratocyst and dentigerous cyst

Haragannavar VC, Tegginamani AS, Raju S, Kudva S, Peter CD, Shruthi DK

Indian J Pathol Microbiol, 2019 2 2;62(1):3-6.
PMID: 30706851 DOI: 10.4103/IJPM.IJPM_403_18

Background: FHIT (Fragile histidine triad) a member of tumor suppressor family, has been extensively studied in many solid tumors including head and neck squamous cell carcinoma. Among all head and neck cyst and tumors odontogenic lesions account approximately 3%-9%. The molecular pathogenesis of these lesions is less explored. Defects in cell cycle regulators and tumor suppressor genes could result in the development of odontogenic cyst and tumors. Hence, we aimed to determine the significant role of a tumor suppressor gene FHIT in most commonly occurring odontogenic lesions mainly ameloblastoma, odontogenic keratocyst and dentigerous cyst.
Subjects and Methods: Immunohistochemical analysis of FHIT was done in ameloblastoma, odontogenic keratocyst, dentigerous cyst and dental follicle. Interpretation of the stained slides were done using standard scoring criteria by two pathologist. The results were subjected for statistical analysis.
Results: Expression of FHIT varied among the groups, with highest negative expression in ameloblastoma 44.4% followed by odontogenic keratocyst 14% and 100%positive expression was seen in dentigerous cyst. The expression levels between the groups were statistically insignificant.
Conclusion: The varied expression or negative expression of FHIT could be considered as an indicator for aggressive behavior and transformation of preneoplastic/cystic epithelium.

Matched MeSH terms: Ameloblastoma/genetics*
Epithelial-to-mesenchymal transition in ameloblastoma: focus on morphologically evident mesenchymal phenotypic transition

Siar CH, Ng KH

Pathology, 2019 Aug;51(5):494-501.
PMID: 31262562 DOI: 10.1016/j.pathol.2019.04.004

The ameloblastoma is the most common and clinically significant odontogenic epithelial neoplasm known for its locally-invasive behaviour and high recurrence risk. Epithelial-to-mesenchymal transition (EMT) is a fundamental process whereby epithelial cells lose their epithelial characteristics and gain mesenchymal properties. EMT induction via transcription repression has been investigated in ameloblastoma. However, morphologically evident mesenchymal phenotypic transition remains ill-defined. To determine this, 24 unicystic (UA), 34 solid/multicystic (SA) and 18 recurrent ameloblastoma (RA) were immunohistochemically examined for three EMT-related mesenchymal markers, alpha smooth muscle actin (α-SMA), osteonectin and neuronal cadherin (N-cadherin). All three factors were heterogeneously detected in ameloblastoma samples (α-SMA, n=71/76, 93.4%; osteonectin, n=72/76, 94.7%; N-cadherin, n=24/76, 31.6%). In the tumoural parenchyma, immunoreactive cells were not morphologically distinct from their non-reactive cellular counterparts. Rather, α-SMA and osteonectin predominantly labelled the cytoplasm of central polyhedral > peripheral columnar/cuboidal tumour cells. N-cadherin demonstrated weak-to-moderate circumferential membranous staining in both neoplastic cell types and cytoplasmic expression in spindle-celled epithelium of desmoplastic amelobastoma. For all tumour subsets, α-SMA and osteonectin scored significantly higher in the stroma > parenchyma whilst α-SMA was overexpressed along the tumour invasive front > centre (p<0.05). Stromal N-cadherin scored higher in SA > UA and RA > UA (p<0.05). Other clinicopathological parameters showed no significant associations. Taken together, acquisition of mesenchymal traits without morphologically evident mesenchymal alteration suggests partial EMT in ameloblastoma. Stromal upregulation of these proteins in SA and RA implicates a role in local invasiveness.

Matched MeSH terms: Ameloblastoma/pathology*
Parenchyma-stromal interleukin-1 alpha and interleukin-6 overexpressions in ameloblastoma correlate with the aggressive phenotype

Goh YC, Chan SW, Siar CH

Malays J Pathol, 2019 Dec;41(3):303-311.
PMID: 31901915

INTRODUCTION: Ameloblastoma is a benign but locally invasive odontogenic epithelial neoplasm with a high recurrence rate after treatment. The two main subsets encountered clinically are unicystic (UA) and solid/multicystic ameloblastoma (SMA). Currently neoplastic progression of many tumour types are believed to be related to parenchyma-stromal cell-cell interactions mediated by cytokines notably interleukins (IL). However their roles in ameloblastoma remain ill-understood.
MATERIALS AND METHODS: Thirty-nine formalin-fixed paraffin-embedded ameloblastoma cases comprising unicystic ameloblastoma (n=19) and solid/multicystic ameloblastoma (n=20) were subjected to IHC staining for IL-1α, IL-1β, IL-6 and IL-8. A semi-quantitative method was used to evaluate the expression levels of these cytokines according to cell types in the tumoural parenchyma and stroma.
RESULTS: Major findings were upregulations of IL-1α and IL-6 in SMA compared to UA. Both cytokines were heterogeneously detected in the tumoural parenchyma and stroma. Within the neoplastic epithelial compartment, IL-1α expression was more frequently detected in PA-like cells in UA whereas it was more frequently encountered in SR-like cells in SMA. IL-6 demonstrated higher expression levels in the stromal compartment of SMA. IL-1β and IL-8 were markedly underexpressed in both tumour subsets.
CONCLUSIONS: Overexpression of IL-1α in SMA suggests that this growth factor might play a role in promoting bone resorption and local invasiveness in this subtype. The expression levels of IL-1α and IL-6 in three cellular localizations indicate that parenchymal-stromal components of ameloblastoma interact reciprocally via IL-1α and IL-6 to create a microenvironment conducive for tumour progression.

Matched MeSH terms: Ameloblastoma/metabolism*
Aggressive (malignant?) epithelial odontogenic ghost cell tumour of the maxilla

Siar CH, Ng KH

J Laryngol Otol, 1994 Mar;108(3):269-71.
PMID: 8169519

A case is described of ameloblastoma of the mandible presenting with multiple recurrences and subsequent extension to the maxilla with resultant transformation into an aggressive (malignant?) epithelial odontogenic ghost cell tumour. The latter is a rare, biologically virulent entity that affects mainly males, exhibits a preference for the maxilla and is histologically characterized by atypical malignant odontogenic epithelium associated with areas of ghost cell formation and varying amounts of dentinoid.

Matched MeSH terms: Ameloblastoma/pathology*
Desmoplastic variant of ameloblastoma in Malaysians

Ng KH, Siar CH

Br J Oral Maxillofac Surg, 1993 Oct;31(5):299-303.
PMID: 8218083

Seventeen cases are reported of desmoplastic variant of ameloblastoma of the jaws observed during the years 1967-1991. There were 12 females and 5 males, and these consisted of 7 Chinese, 6 Malays, 2 Indians, 1 Sikh and 1 Kadazan. Their ages at diagnosis ranged from 21-60 years with a mean of 36.6 years. There were 10 mandibular and 7 maxillary tumours. Of these, 14 cases involved the anterior segment with extension to the premolar region in 5 cases. 60% of cases were radiologically suggestive of fibro-osseous lesions. The main mode of treatment was resection and 1 case presented with recurrence. The findings of this study were compared with those of previous reports.

Matched MeSH terms: Ameloblastoma/pathology*
Mandibular reconstruction: experience with the free vascularized fibula transfer

Khanijow VK, Ahmad TS, Lian CB, Jalaludin MA

Microsurgery, 1993;14(6):375-9.
PMID: 8371683

Mandibular resection, following surgery for tumor or osteoradionecrosis, leaves a patient with a swallowing, speech, and cosmetic disability. Repair of the oromandibular defect is difficult and various prostheses and grafts have been used and reported. The most popular form of mandibular reconstruction is the use of the free, vascularized bone transfer. We report our experience with the free vascularized fibula bone transfer in eight patients.

Matched MeSH terms: Ameloblastoma/surgery

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