Cases Of Adamantinoma Research Articles

Adamantinoma: About 4 Cases

Adamantinoma is a rare primary malignant bone tumor of the tibia in young adults. Histologically, it comprises malignant epithelial cells within a fibrous or osteofibrous stroma. Adamantinoma poses a problem of differential diagnosis with osteofibrous dysplasia and osteofibrodysplasia. the evolution of this low-grade sarcoma is slow. Treatment is surgical, involving resection of the tumour and reconstruction of the bone. Metastases may occur in the lungs, lymph nodes or bones. We report 4 cases of adamantinoma.

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.

Clinicopathological observation of adamantinoma of long bone

Objective: To investigate the clinicopathological features and differential diagnosis of adamantinoma of long bone. Methods: Seven cases of adamantinoma on long bone were selected at Jiangsu Province People's Hospital from June 2012 to May 2018. Clinicopathologic details, immunohistochemical and molecular analysis were performed,and the relevant literature reviewed. Results: There were 6 males and 1 female patients,age ranging from 21 to 60 years (mean 38 years). Six cases were on the right side and one case was on the left; in five cases the tumors arose from tibia, one from patella and one from humerus. Microscopically,tumour cells were mainly composed of spindle cells arranged in bundles or braids,with irregular epithelial island. Immunohistochemically,the epithelial island expressed high molecular weight cytokeratin but not CK8/18. Both epithelial and spindle components expressed vimentin. One case that was microscopically similar to intraosseous synovial sarcoma did not show SYT gene rearrangement. Clinical follow-up was available for five patients: one patient had axillary metastases seven months after operation, one patient had recurrence 34 months after surgery, 3 patients were uneventful with follow up duration from half a month to 32 months. Conclusion: Adamantinoma occurring in long bones is very rare. The correct diagnosis requires adequate sample selection, careful morphologic observation, immunohistochemistry and molecular genetics.

TIBIAL ADAMANTINOMA: ANALYSIS OF SEVEN CONSECUTIVE CASES IN A SINGLE INSTITUTION.

ABSTRACTObjective:Adamantinoma accounts for less than 1% of the primary bone neoplasms. The tibia is the most affected bone and it is predominant in male patients between the second and third decades of life. The objective of this study is to obtain epidemiological and clinical information on patients with adamantinoma of the tibia treated surgically between 1989 and 2016.Methods:Retrospective series of seven patients diagnosed with adamantinoma of the tibia that underwent surgery at the orthopedic oncology service of our hospital. The information was obtained from the medical records and histopathological reports of our institution.Results:A total of 2870 medical records with histological reports were evaluated. Seven cases of adamantinoma of the tibia were included. The mean age was 28.5 (17-49) years. We found a predominance of females (71.4%) and the most affected side was the left one, with four cases (57.1%). The biopsy revealed bone adamantinoma in four (57.1%) patients, while the diagnosis of the other patients was confirmed after the histological examination of the surgical specimen. All the patients underwent surgery as definitive treatment. No positive margins were reported. No local recurrence (LR) was reported and two patients had distant metastasis (DM).Conclusion:The prognosis of survival in cases of adamantinoma of the tibia is high. The rates of LR and DM were low. Surgical treatment with extensive tumor resection is the treatment of choice. Level of Evidence IV, Case Series.

Mesenchymal–Epithelial Differentiation of Adamantinoma of Long Bones: An Immunohistochemical and Ultrastructural Study

Three cases of adamantinoma were studied by electron microscopy and immunohistochemistry. In the tubular pattern, well-differentiated epithelial cells and glandular structures were present, in addition to ill-defined glands. In the basaloid pattern, less differentiated epithelial cells with discohesion were seen in the central epithelial masses. This study established the epithelial nature of some tubular structures with slit-like lumina, easily misinterpreted as capillaries by light microscopy. Results also showed that the irregular spaces observed within the basaloid pattern probably result from cell discohesion. Moreover, this investigation demonstrates the epithelial nature of a subset of spindle cells within the stroma of adamantinoma and offers ultrastructural evidence for a probable mesenchymal–epithelial transformation as its histogenesis.

P63 expression in adamantinoma

Adamantinoma is a rare primary bone neoplasm with epithelial differentiation that is frequently associated with a concomitant fibrous component. Clinical, cytogenetic and histomorphologic overlap has previously been described with osteofibrous dysplasia, thereby suggesting a relationship between these two lesions. We performed a retrospective review of our archives to characterize the clinical and pathologic aspects of adamantinoma and osteofibrous dysplasia diagnosed at our institution, and to compare the expression patterns of p63 and keratin. Nine cases of adamantinoma (six classical, three osteofibrous dysplasia-like) and 11 cases of osteofibrous dysplasia were identified. The epithelial component in adamantinoma was found to stain for p63. Rare cells expressing p63 were also identified in eight cases of osteofibrous dysplasia. Expression of p63 was not identified in any of the five cases of fibrous dysplasia controls. The presence of staining for p63, albeit rare, in osteofibrous dysplasia supports the notion of a possible relationship between osteofibrous dysplasia and adamantinoma. Furthermore, our results suggest that, in some situations, p63 may be useful in helping differentiate metastatic carcinoma from 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 long bones. The experience at the Istituto Ortopedico Rizzoli.

All cases of adamantinoma seen at the Istituto Ortopedico Rizzoli were retrospectively reviewed. Although this tumor is exceedingly rare, nine cases were collected. The tumor is composed of four histological patterns: spindle, basaloid, squamoid, and tubular. The prognosis of this tumor depends on the adequacy of therapy. Surgery that is expedient and adequate tends to offer the best prognosis.

Department of Radiodontia: Adamantine Tumors of the Jaw

EIGHT cases of adamantinoma have been admitted to the New York State Institute for the Study of Malignant Disease. During this period, a total of 4,360 tumor cases of all descriptions were accepted for treatment, thus making the ratio of adamantine tumors approximately 1 to 545, or about one-fifth of 1 per cent of all tumors seen at this Institute. Although this type of tumor is not rare, it seems justifiable to report the following cases. Case 1. No. 5,499, admitted October 29, 1917, male, married, 38 years of age. Family history unimportant. In 1913, the patient first noticed a soreness in the right lower jaw. He had a tooth extracted, believing this would put an end to the trouble. The soreness continued and, in 1916, he had three more teeth removed. At the time of admission there was a large lump in the lower jaw, but no history of “lumps” in the neck. Expectoration was excessive and there was a slight odor. The patient did not complain of pain. Examination disclosed a well-nourished man, physically n...