Adamantinoma Research Articles

Adamantinoma: A SEER-based Epidemiological Analysis

Adamantinoma (AD) is a rare bone cancer accounting for less than 0.1–0.5% of all primary bone tumors. No consensus guidelines exist for the treatment of this disease and long-term (twenty-year) survival has yet to be explored. The Surveillance, Epidemiology, and End Results (SEER) Program was queried for patients with a diagnosis of primary AD (ICD-O-3 code 9261/3). Demographic and treatment variables were analyzed via Fisher’s Exact Test and 20-year overall survival (20y OS) was assessed via log-rank analysis. Seventy-four patients with AD were identified; median age was 20–24 years. Multivariate analysis demonstrated that patients < 25 years of age at diagnosis with AD had increased 20y OS compared to those > 24 years (HR = 0.28; p = 0.028), while no other variables influenced survival. Subanalysis demonstrated patients > 40 years saw decreased survival (46% [11%, 81%]) compared to those ≤ 40 years (96% [89%, 104%]; p = 0.005). Patients ≤ 40 years of age at diagnosis were more likely to have local disease (78% of all 49 local cases) and less likely to have distant disease (0% of two cases) compared to patients > 40 years (p = 0.017). Stratifying by surgical procedure, no difference in 20y OS was appreciated (p = 0.12). Younger age at diagnosis provides mortality benefit and increased proportion of localized disease for those diagnosed with AD. No other demographic or treatment variables were found to influence 20y OS. Population-based analysis of AD is limited both by disease rarity and incomplete coding within SEER.

Surgical Outcome and Oncological Survival of Osteofibrous Dysplasia-Like and Classic Adamantinomas: An International Multicenter Study of 318 Cases.

Osteofibrous dysplasia-like adamantinoma (OFD-AD) and classic adamantinoma (AD) are rare, neoplastic diseases with only limited data supporting current treatment protocols. We believe that our retrospective multicenter cohort study is the largest analysis of patients with adamantinoma to date. The primary purpose of this study was to describe the disease characteristics and evaluate the oncological outcomes. The secondary purpose was to identify risk factors for local recurrence after surgical treatment and propose treatment guidelines. Three hundred and eighteen confirmed cases of OFD-AD and AD for which primary treatment was carried out between 1985 and 2015 were submitted by 22 tertiary bone tumor centers. Proposed clinical risk factors for local recurrence such as size, type, and margins were analyzed using univariable and multivariate Cox regression analysis. Of the 318 cases, 128 were OFD-AD and 190 were AD. The mean age at diagnosis was 17 years (median, 14.5 years) for OFD-AD and 32 years (median, 28 years) for AD; 53% of the patients were female. The mean tumor size in the OFD-AD and AD groups combined was 7.8 cm, measured histologically. Sixteen percent of the patients sustained a pathological fracture prior to treatment. Local recurrence was recorded in 22% of the OFD-AD cases and 24% of the AD cases. None of the recurrences in the OFD-AD group progressed to AD. Metastatic disease was found in 18% of the AD cases and fatal disease, in 11% of the AD cases. No metastatic or fatal disease was reported in the OFD-AD group. Multivariate Cox regression analysis demonstrated that uncontaminated resection margins (hazard ratio [HR] = 0.164, 95% confidence interval [CI] = 0.092 to 0.290, p < 0.001), pathological fracture (HR = 1.968, 95% CI = 1.076 to 3.600, p = 0.028), and sex (female versus male: HR = 0.535, 95% CI = 0.300 to 0.952, p = 0.033) impacted the risk of local recurrence. OFD-AD and AD are parts of a disease spectrum but should be regarded as different entities. Our results support reclassification of OFD-AD into the intermediate locally aggressive category, based on the local recurrence rate of 22% and absence of metastases. In our study, metastatic disease was restricted to the AD group (an 18% rate). We advocate wide resection with uncontaminated margins including bone and involved periosteum for both OFD-AD and AD. Prognostic Level IV. See Instructions for Authors for a complete description of levels of evidence.

Outcome of osteofibrous dysplasia-like versus classic adamantinoma of long bones: a single-institution experience

BackgroundThe clinical and molecular characteristics of osteofibrous dysplasia (OFD)-like adamantinoma (AD) differ from those of classic AD. Most reports about OFD-like AD are case reports or small case series. More cases from different centers are still warranted.MethodsThe orthopedic oncology database of our institution was searched to identify patients with AD. The cases of OFD-like and classic AD of the long bones were retrospectively analyzed. Between December 1999 and August 2016, 23 patients were treated for AD, comprising seven with OFD-like AD and 16 with classic AD. The outcomes were compared between AD subtypes.ResultsIn the OFD-like AD group, four lesions were treated with extensive curettage, while three were treated with wide resection. The median follow-up duration in the OFD-like AD group was 66 months (range 43–131 months). At the end of follow-up, there was only one case of local recurrence (LR) in the OFD-like AD group, giving a LR rate of 14.3% (1/7). No distant metastasis or progression to classic AD was detected in the OFD-like AD group. In the classic AD group, the treatments were below-the-knee amputation in one patient with extensive tibial and fibular lesions, curettage with a bone graft in one patient who was diagnosed with OFD based on a core needle biopsy, hemi-cortical excision and reconstruction in two patients, and segmental resection and reconstruction in 12 patients. At the end of follow-up, there were three cases of LR in the classic AD group, giving a LR rate of 18.8% (3/16); two patients developed lung metastasis after LR and died of the disease at 88 and 126 months after the first surgery in our hospital, respectively. The classic AD group had a metastatic rate of 12.5% (2/16), a final limb salvage rate of 75%, and estimated 5- and 10-year survival rates of 88.9% and 77.1%, respectively.ConclusionsOFD-like AD has a better outcome than classic AD. For OFD-like AD, extensive curettage is suggested if the tumor extent allows. For classic AD, aggressive resection with wide margins is essential to achieve local control. A long-term follow-up is necessary due to the possibility of late complications.

18F-FDG PET/CT in staging and follow-up of adamantinoma

18F-FDG PET/CT in staging and follow-up of adamantinoma

Chondroblastoma with adamantinoma differentiation of the calcaneus

Chondroblastoma with adamantinoma differentiation of the calcaneus

Osteofibrous Dysplasia and Adamantinoma

Osteofibrous dysplasia (OFD) is a rare, benign, fibro-osseous lesion that typically is seen within the cortex of the tibia in children. Adamantinoma (AD) is a rare, low-grade malignant primary bone tumor that occurs most often in the tibia and/or fibula of adolescent persons and young adults; however, it has been reported in other long bones, as well. Immunohistochemical and ultrastructural evidence has shown that the neoplastic cell in AD derives from an epithelial lineage. More recently, published reports have described another clinical entity-differentiated or OFD-like AD-that appears to lie between OFD and AD along a spectrum of disease. Controversy exists as to whether OFD is a precursor lesion to AD or whether OFD may be a residual lesion resulting from a spontaneously regressing AD. Management of OFD varies from observation to surgical intervention, depending on the age of the patient and the extent of the lesion. Management of AD requires surgical resection with wide margins, followed by appropriate reconstruction, to minimize the risk of local recurrence or metastasis.

Fibular osteoadiposal flap for treatment of tibial adamantinoma: a case report.

We treated a case with left tibial adamantinoma by use of a contralateral fibular osteoadiposal flap. The donor site of conventional fibular osteocutaneous flap must be covered with a skin graft because if we close the donor skin defect directly, compartment syndrome might occur. We were able to close the donor skin defect because this combined type flap included only a small monitoring skin paddle. We present herein the utility of the osteoadiposal flap and show the value of a skin-sparing approach with a minimal aesthetic defect.

Adamantinoma of the tibia; report of a case.

Adamantinoma of the tibia; report of a case.

Controversy about jaws adamantinoma

Controversy about jaws adamantinoma

Expression of cytokeratin 1, 5, 14, 19 and transforming growth factors-beta1, beta2, beta3 in osteofibrous dysplasia and adamantinoma: A possible association of transforming growth factor-beta with basal cell phenotype promotion.

To elucidate the precise origin and characteristics of the epithelial components of osteofibrous dysplasia (OF) and adamantinoma (AD), the expression of transforming growth factor (TGF)-beta1, beta2 and beta3 and cytokeratin (CK) subtypes were studied in five cases of AD and 18 cases of OF by immunohistochemistry. CK1 was expressed in 10 out of 18 OF cases; CK5 was expressed in one OF case; CK14 was positively stained in 10 cases of OF; CK19 was positively stained in 16 OF cases; CK1 was expressed in three out of five AD cases; CK5 was expressed in one case of AD; CK14 was positively stained in four AD cases; and CK19 was positively stained in five AD cases. In OF, TGF-beta1, beta2 and beta3 were expressed in both fibroblasts and osteoblasts. In AD, TGF-beta1, beta2 and beta3 were expressed in both epithelial and fibrous components. These results suggest that epithelial components of AD and OF share epidermal characteristics, CK1, express basal cell phenotype and cytokeratins 5, 14 and 19. In addition to these epithelial characteristics, strong immunoreactivity for TGF-beta poses the possibility of TGF-beta promotion of basal cell phenotype expression for the epithelial components in OF and AD.

Adamantinoma of the jaw

Adamantinoma of the jaw

So-Called Adamantinoma of the Tibia (Two Cases)

So-Called Adamantinoma of the Tibia (Two Cases)

A Cace of Adamantinoma, Suggestive of Epithelioma, of Mandibula

A Cace of Adamantinoma, Suggestive of Epithelioma, of Mandibula

Extensive Adamantinoma of Mandible

Extensive Adamantinoma of Mandible

Adamantinoma of the mandible: analysis of surgical treatment.

Adamantinoma of the mandible: analysis of surgical treatment.

Considerations about hard and dentified adamantinoma: M. Dechaume and B. Kerebel. Rev. de stomatol. 53: 885, 1953

Considerations about hard and dentified adamantinoma: M. Dechaume and B. Kerebel. Rev. de stomatol. 53: 885, 1953

A contribution to the knowledge of the histology, histogenesis and etiology of adamantinomas.

A contribution to the knowledge of the histology, histogenesis and etiology of adamantinomas.

Adamantinomas in relation to the nose: Ahmad Bey Handousa. J. Laryng. & Otol. 65: 715, 1951

Adamantinomas in relation to the nose: Ahmad Bey Handousa. J. Laryng. & Otol. 65: 715, 1951

Melanotic adamantinoma of mandible in child: S. M. Wass, M.S., F.R.C.S., Proc. Roy. Soc. Med. 41: 281, 1948

Melanotic adamantinoma of mandible in child: S. M. Wass, M.S., F.R.C.S., Proc. Roy. Soc. Med. 41: 281, 1948

Melanotic Adamantinoma of the Mandible in a Child Aged 5 Months

Melanotic Adamantinoma of the Mandible in a Child Aged 5 Months