Ameloblastic carcinoma with features of ghost cell odontogenic carcinoma in a patient with suspected Gardner syndrome

Fitzpatrick, Sarah; Hirsch, Stanley; Listinsky, Catherine M.; Lyu, Dan; Baur, Dale A.

doi:10.1016/j.oooo.2014.09.028

Cited by 17 publications

(15 citation statements)

References 18 publications

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“…From our perspective, the histological spectra of odontogenic carcinoma with dentinoid, AMECA with ghost cells, CCOC with dentinoid and CCOC with ghost cells are within the histological spectrum of AMECA. In this context, we support the idea that it is still premature to separate such entities as distinct nosological entities …”

Section: Discussionmentioning

confidence: 86%

Ameloblastic carcinoma: a Brazilian collaborative study of 17 cases

et al. 2016

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Section: Discussionmentioning

confidence: 86%

Ameloblastic carcinoma: a Brazilian collaborative study of 17 cases

et al. 2016

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“…In both cases the clinical and radiological features were consistent with a dentigerous cyst [12,13]. Fitzpatrick et al [11] reported of a tumor combining, predominantly, characteristics of ameloblastic carcinoma and ghost cell odontogenic carcinoma in a 37-year-old male African American. The patient exhibited numerous dental abnormalities in addition to osteomas of the mandible, therefore, GS was strongly suspected but could not be confirmed [11].…”

Section: Discussionmentioning

confidence: 64%

“…Dental anomalies, odontomas, supernumerary and impacted teeth, are frequently encountered in GS [2,3]. With the exception of dentigerous cysts, other types of odontogenic cysts and neoplasms are scarce in patients with GS [11]. Patel and Rees [12] and Bates et al [13] presented one case each of unicystic ameloblastoma affecting a 14-year-old male and a 15-year-old female patient, respectively.…”

Section: Discussionmentioning

confidence: 99%

“…Fitzpatrick et al [11] reported of a tumor combining, predominantly, characteristics of ameloblastic carcinoma and ghost cell odontogenic carcinoma in a 37-year-old male African American. The patient exhibited numerous dental abnormalities in addition to osteomas of the mandible, therefore, GS was strongly suspected but could not be confirmed [11]. A collective presentation of the clinico-pathologic characteristics of previously reported cases of odontogenic neoplasms associated with GS is provided in Table 2.…”

Section: Discussionmentioning

confidence: 99%

“…A review of the English literature revealed only three cases of odontogenic neoplasms developing in patients with GS, specifically two unicystic ameloblastomas [12,13] and a case of ameloblastic carcinoma [11].…”

Section: Introductionmentioning

confidence: 99%

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Orthokeratinized Odontogenic Cyst with an Associated Keratocystic Odontogenic Tumor Component and Ghost Cell Keratinization and Calcifications in a Patient with Gardner Syndrome

Argyris

Koutlas

2016

Head and Neck Pathol

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Gardner syndrome (GS) is caused by mutations in the APC and besides adenomatous colorectal polyps includes such manifestations as osteomas, epidermoid cysts (ECs) and occasionally multiple pilomatricomas. More than 50 % of ECs in patients with GS exhibit pilomatricoma-like ghost cell keratinization. The latter may be explained by the fact that the development of both GS and pilomatricoma is driven by activation of the Wnt/b-catenin signaling pathway. A 62-year-old, Caucasian male with history of GS presented with a unilocular, mixed radiopaque/radiolucent mandibular lesion causing divergence and external root resorption of involved teeth. Histopathologically, the lesion was composed of two cystic components, an orthokeratinized odontogenic cyst (OOC) and a smaller one with characteristics of keratocystic odontogenic tumor (KCOT) featuring, focally, ghost cells and an epithelial morule-like structure. Dystrophic calcifications essentially similar to those seen in pilomatricomas were observed in the fibrous connective tissue wall. The KCOT and OOC epithelia revealed strong and diffuse cytokeratin (AE1/AE3) and b-catenin immunoreactivity. CD10 positive immunostaining was seen in the keratin and superficial spinous cell layers in both OOC and KCOT. The intraepithelial and mural ghost cells showed a cytokeratin (?), b-catenin and CD10 (-) immunophenotype. The diagnosis of OOC with ghost cell calcifications in association with KCOT was rendered. The patient was lost to follow-up. Although a coincidental co-existence cannot be excluded, ghost cell calcifications mimicking pilomatricoma-like changes in an unusual odontogenic cyst combining OOC and KCOT features as seen in this patient with GS may be explained by the common molecular mechanisms underlying the pathogenesis of cutaneous pilomatricomas and GS.

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