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At least 19 recordsLinked to original sources
PubMed2026

Genomic Characterization of Classic Adamantinoma, Osteofibrous Dysplasia, and Osteofibrous Dysplasia-like Adamantinoma.

Classic adamantinoma, osteofibrous dysplasia (OFD), and OFD-like adamantinoma are rare bone tumors arising primarily in the tibiae. Their distinction can be challenging; data on their molecular pathogenesis remain limited. We searched our pathology files in 2004-2024 for available cases and performed targeted next-generation sequencing along with whole-genome single-nucleotide polymorphism arrays and 3-dimensional genomics/Hi-C sequencing in selected cases. Our cohort included 3 classic adamantinomas (2 females and 1 male; age, 14-56 years), 5 OFDs (3 females and 2 males; age, 9-25 years), and 2 OFD-like adamantinomas (1 female and 1 male; age, 30-41 years). Of the 10 tumors, 9 arose from the tibiae; 1 classic adamantinoma originated from the radius. The 3 classic adamantinomas harbored multiple copy number gains involving chromosome 7, 8, 10, 12, and/or 19. Focal deletion of chromosome 17, intergenic rearrangement involving FGFR1, and NRAS p.G12D were each present in 1 classic adamantinoma. Of the 5 OFDs, KMT2A p.C2441F, KMT2D p.S1040P, PHOX2B p.G213D, and RIF1 deletion were each present in 1 case; no additional copy number/single-nucleotide variants were identified. Of the 2 OFD-like adamantinomas, one case with tumor clusters visible only on cytokeratin immunostain harbored no variants, whereas another case with tumor clusters visible on light microscopy and cytokeratin/p40 immunostains showed gains of chromosome 7, 8, 19, and 20. By Hi-C, 1 classic adamantinoma harbored an approximately 9 Mb tandem duplication on chromosome 12q, 1 OFD harbored a rearrangement with breakpoints near MECOM and HOOK3, and the OFD-like adamantinoma with tumor clusters visible only on cytokeratin immunostain harbored no structural variant. In conclusion, classic adamantinomas and OFD might be genetically distinct. Classic adamantinomas harbored multiple alterations, including chromosome/arm-level copy number gains, the detection of which could aid their distinction from OFDs. Using genomics as the benchmark, OFD-like adamantinomas might be better delineated by light microscopy or p40 than by cytokeratin immunohistochemistry. These data expanded our molecular understanding of these rare bone tumors.

Humans↗
PubMed1978

["Adamantinoma" of the tibia and reactive bone changes (author's transl)].

A case of "adamantinoma" of the tibia is reported. 10 years ago a giant tumor with fibrous dysplasia had been diagnosed in a biopsy taken from the same site of cystic bone changes. These findings may occur as reaction in the periphery of "adamantinomas" of long bones. By this experience the importance was emphasized, to obtain tissue from the center portion of the tumor to include the typical epithelial islets pathognomonic for "adamantinomas", which in our case, were found only 10 years after the first operation. Our patient was treated by curetting and filling of the defect with bony splinters. At this time, 8 months after surgery, he is without complaints and in full use of his diseased leg.

Ameloblastoma↗
PubMed1978

Ultrastructural findings supporting the angioblastic nature of the so-called adamantinoma of the tibia.

Electron microscopy of a case of adamantinoma of the tibia shows features which support a mesenchymal angioblastic origin rather than epithelial. Comparison is made between tissue from this tumour and a squamous cell carcinoma of the femur arising in chronic osteomyelitis. In adamantinoma no desmosomes are found between tumour cells and their cytoplasmic ultrastructure shows features of mesenchymal cell type including evidence of pinocytic activity and bundles of filaments resembling hyperplastic endothelial cells. The stroma shows features similar to fibrous dysplasia of bone with fibroblasts, histiocytes and fibrolipoblastic lipid-laden mesenchymal cells. It is condluded that adamantinoma of the tibia should be considered to be an angioblastic tumour of bone.

Bone Neoplasms↗
PubMed1979

[Differential diagnosis of a metastasizing adamantinoma of the tibia and fibula (author's transl)].

A case of an adamantinoma of the tibia and fibula is described in a 10-year-old girl who died after 17 years with pulmonary metastasis. For a long time, an exact diagnosis couldn't be made. Histologically the lesion was called chronic osteomyelitis (Brodie's abscess), synovial sarcoma and spindle cell sarcoma. The revision of the histological sections revealed a fibrous dysplasia-like pattern in the first lesion of the tibia with tiny foci of an epitheloid-like pattern. This had could be the key to the right diagnosis, because sometimes adamantinoma of long bones is associated with a fibrous dysplasia-like pattern. The reason for misinterpretation of the histological features is seen in the typical variable histological pattern of this tumor (basaloids, squamoid, spindled and tubular pattern). In this connection is refered to the necessity to correlate the histological feature with the X-ray in making diagnosis in bone pathology.

Adult↗
PubMed1978

Ossifying fibroma of long bone: its distinction from fibrous dysplasia and its association with adamantinoma of long bone.

Two cases of ossifying fibroma of long bones are presented. This tumor is confused with monostotic fibrous dysplasia, but can be distinguished by its intracortical location, as demonstrated radiographically, and by its histologic pattern. Distinction from fibrous dysplasia is important since ossifying fibroma of long bone is a more aggressive lesion with different therapeutic implications. It appears that ossifying fibroma and adamantinoma of long bones are somehow related, and that lesions resembling fibrous dysplasia in association with adamantinomas of long bones are actually ossifying fibromas.

Adolescent↗
PubMed1976

[A so-called adamantinoma of the right femur (author's transl)].

Report on a so-called adamantinoma of a right femur (fourth case from available publications) with remarks on the histogenesis of these tumors, which show correlating properties with malignant synovialomas. In other words, the so-called adamantinoma of long bones represents the osseous form of a malignant synovialoma, whereby an intraosseous ganglion has to be considered as a histogenetic criterion (matrix).

Diagnosis, Differential↗
PubMed1977

Tibial adamantinoma: its histogenesis from ultrastructural studies.

Only three previous reports on the ultrastructural appearance of tibial adamantinoma have been found among some 120 cases of this tumor reported in the literature. The tumor in our case is less differentiated than the cases previously reported. We believe it is of epithelial character and the possibilities of its histogenesis have been narrowed down to either mesothelial, synovial or squamous origins among the many theories proposed. Ameloblastic origin, however, cannot be ruled out.

Adult↗
PubMed1979

Malignant tumor of humerus with features of "adamantinoma" and Ewing's sarcoma.

This female patient was born in 1935 and died in 1976 with a malignant bone tumor involving the proximal humerus following multiple local recurrences, axillary lymph node metastases, and pulmonary metastases. Histologically, over the course of time, there was an increase in features of an atypical Ewing's sarcoma, at the expense of findings of a typical, extra-cranial so-called adamantinoma. In contrast to another report, our case showed neither epithelial nor endothelial features on fine structural examination, but rather primitive mesenchymal cells, as is described for Ewing's sarcomas.

Adult↗
PubMed1979

Adamantinoma tibiae.

A case with characteristic roentgenographic and microscopic findings of adamantinoma tibiae is reported. Knee disarticulation with early ambulation is the treatment of choice and was performed on this patient. Limb ablation followed by Unna Paste Bandage to prevent oedema and permit early prosthetic fitting facilitates rehabilitation. The 1 year follow-up showed excellent prosthetic function and no evidence of residual tumour.

Adult↗
PubMed1979

Adamantinoma of the appendicular skeleton.

Adamantinoma is a slowly developing tumor originating in the bones of the extremities and containing cells that resemble amelioblasts but do not produce enamel. Anamantinoma's clinically and histologically benign appearance is deceptive, for the recurrence rate is high unless surgical extirpation is complete. Thus, aggressive surgical excision of the involved area of bone should be performed at the time of diagnosis. Allograft substitution may be an effective alternative to amputation.

Adolescent↗
PubMedDate not supplied

[Histomorphological features of adamantinoma].

The histomorphological features of ameloblastoma and the literature classifications of this form are examined and a personal series of 19 cases involving the mandible and 2 involving the upper jaw is presented. Cord-like and plexiform pictures were followed by follicular, cystic, cylindromatous and stellate forms in order of frequency. Death occurred in two cases of cylindromatous ameloblastoma.

Aged↗