Adamantinoma of Long Bones: A Histopathological and Immunohistochemical Study of 23 Cases

doi:10.1016/S0344-0338(11)80560-1

Pathology - Research and Practice

Volume 191, Issue 2, March 1995, Pages 112-120

https://doi.org/10.1016/S0344-0338(11)80560-1 Get rights and content

Summary

The clinical and histological data of twenty-three cases of adamantinomas of the long bones collected by the Working Group on Bone Tumors at the DKFZ/FRG are reported including immunohistochemical observations in twenty-one of the cases. Females and males between 5 and 67 years (mean, 25.4 years) were affected equally (11/12). All adamantinomas were positive for cytokeratins often in coexpression with vimentin, at least focally. Although exhibiting varying histological patterns, no correlation between histology and clinical course was seen. However, sex and mode of initial therapy seem to influence an unfavorable clinical outcome. All three deceased patients were males receiving marginal or delayed surgery. This underlines the low-grade malignant character of adamantinoma. To assure the histological diagnosis pathologists should employ immunohistochemistry for demonstrating the sometimes sparse epithelial cell nests when radiology is suggestive for adamantinoma. Correct diagnosis should lead to resection with wide surgical margins.

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Cited by (47)

Adamantinoma: A review of the current literature
2023, Journal of Bone Oncology
Adamantinoma is a rare primary low-grade malignant tumour of the appendicular skeleton most commonly found in the tibia. It has an indolent course, with local recurrences and lung metastases occurring over a protracted duration. There have been several suggestions pertaining to a vascular origin in the literature, however, histogenesis remains unclear. Currently, guidelines are not available pertaining to clinical management.
This paper presents an overview of the current literature regarding this unusual malignancy. It also explores disease etiology and acknowledges the benefits and challenges of investigations pertaining to diagnosis. It recognizes a paucity of recommendations regarding appropriate surveillance and follow up. This review aims to assist clinicians in the building of a consensus opinion for optimal adamantinoma case management under current circumstances where formal guidelines do not exist.
Osteofibrous Dysplasia and Adamantinoma
2021, Surgical Pathology Clinics
Citation Excerpt :
Similar to OFD, ADA occurs almost exclusively in the anterior cortex of the diaphysis, and less frequently involves the metaphysis, of the tibia, with a subset of patients also having isolated or concurrent involvement of the fibula.9,20–32 Rarely, this tumor has reportedly arisen in other bones, such as the humerus, ulna, and radius, among others.9,20–32 Patients diagnosed with classic ADA are generally within the third and fourth decades of life, whereas those with OFD-like ADA are younger, often within the same age range as patients with OFD.9
High-grade squamous cell carcinoma arising in a tibial adamantinoma
2019, Human Pathology
Citation Excerpt :
However, metastasis occurs in approximately 25% of cases, often after a significant disease-free interval, reducing the long-term survival rate to approximately 40% at 20 years [10,11]. Younger patient age at diagnosis, pain at presentation, lack of squamous differentiation, intralesional or marginal surgery, and extracompartmental growth are associated with increased likelihood of recurrence and metastasis [5,12,13]. Histologically, adamantinoma is divided into osteofibrous-like adamantinoma and classic adamantinoma.
Adamantinoma of the long bones is a rare, typically low-grade malignant tumor that frequently involves the tibia. Radiographically, adamantinoma is characteristically a lytic, intracortical, and expansile lesion with variable margins. Histologically, adamantinoma is a bimorphic neoplasm, composed of epithelial and osteofibrous elements. Herein, we describe a 72-year-old man with a long-standing tibial mass that, on imaging, rapidly developed cortical destruction with soft tissue extension. Imaging revealed no evidence of a distant site of origin. Needle core biopsy demonstrated high-grade squamous cell carcinoma, and metastasis was initially favored. However, the combined clinicoradiologic and pathologic features were most compatible with a high-grade squamous cell carcinoma arising in adamantinoma. The diagnosis was confirmed in the resection specimen. Both the age at presentation and histologic features make this case unusual and highlight a potential for misdiagnosis in the evaluation of squamous cell carcinoma–containing lesions of the tibia, reinforcing the importance of clinicoradiologic correlation in bone pathology.
Primary lesions that may imitate metastatic tumors histologically: A selective review
2018, Seminars in Diagnostic Pathology
Citation Excerpt :
The histologic appearance of this tumor encompasses collections of spindle cells, aggregates of basaloid cells, and glandular or tubular structures, potentially simulating metastatic carcinoma. The first of those patterns may also imitate a fibroblastic or fibrohistiocytic proliferation.250 Irrespective of their cytological appearances, ADAs are immunoreactive for pankeratin and may also express epithelial membrane antigen, p63, or carcinoembryonic antigen.247,248,251
Several primary pathologic entities in diverse anatomic locations have the potential to simulate metastatic neoplasms histologically. Their misinterpretation as such may result in needless and extensive clinical evaluations that are intended to detect a presumed malignancy at another site. More importantly, mistakes of this type can deprive patients of surgical excisions that could be curative. This presentation considers a review of selected primary lesions that can simulate metastases. They include hemangioblastoma, glioblastoma and meningioma with epithelial metaplasia, choroid plexus carcinomas, primary neuroendocrine carcinomas in unusual locations, special forms of sinonasal and salivary glandular adenocarcinoma, clear-cell thyroid carcinomas, unusual microscopic subtypes of pulmonary adenocarcinoma, epithelioid myomelanocytomas (“sugar tumors”), mesotheliomas, primary thymic carcinomas, endodermal choristomas of the interatrial myocardium, peripheral cholangiocarcinoma, adrenocortical carcinoma, adenocarcinomas of the urinary bladder, mucinous and “rhabdoid” tumors of the ovaries, rete testis adenocarcinomas, interdigitating dendritic-cell sarcoma of lymph nodes, selected sweat gland carcinomas, cutaneous Merkel cell carcinoma, primary dermal and subcutaneous melanoma, mucosal and visceral melanomas, epithelioid sarcoma, clear-cell sarcoma, and adamantinoma of long bones. Differential diagnostic observations are emphasized in reference to those lesions.
Long-term outcome following treatment of Adamantinoma and Osteofibrous dysplasia of long bones
2016, Orthopaedics and Traumatology: Surgery and Research
Adamantinoma (AD) is an ultimately rare, low-grade malignant bone tumor. In most cases it occurs in the tibia of young adults. Osteofibrous dysplasia (OFD) is a rare, benign, lesion that is typically seen in children. Histopathology, ultrastructure, and cytogenetics indicate that these lesions are closely related. Yet, etiology remains a matter of debate. Local recurrence rates are high for both entities as published in literature and long-term outcomes are scarce, due to the rarity of the disease.
AD should be treated by En-Bloc resection while ODF can be treated by curettage or by observation. Consequently, the aim of the present study was to answer following questions: Were local recurrence rates of both entities different based on a retrospective review within a tertiary referral center for orthopedic oncology?
In a retrospective cohort study, 10 patients with AD and 5 patients with OFD (including 1 patient with OFD-like-AD) were reviewed. Primary surgeries for patients with AD were: En-bloc resection in 7, curettage in 2 and amputation in 1. In the OFD group, only 2 patients underwent surgery by curettage. Mean follow-up was 16 years (range: 2–47 years). Nine patients had a minimum follow-up of 10 years (mean: 23 years; range: 10–47 years).
Four patients with AD (40%) and 2 patients with OFD (40%) – all of them following surgical removal – suffered from local recurrence. In the “En bloc” resection group of AD, there were 2 LR (29%). All patients of both groups treated with curettage showed LR. One patient with AD had metastasis at time of diagnosis and died of disease. Another patient with AD was diagnosed with metastasis 67 months after surgery and was still alive with disease at latest follow-up (77 month).
The overall prognosis of AD and OFD is good, yet local recurrence rates are high, irrespective of surgical strategy. While an internationally standardized treatment regime is still missing, a more radical surgical approach should be considered, especially when treating AD.
Retrospective study; Level IV.
Benign Tumors and Tumor-Like Conditions of Bone
2014, Pathobiology of Human Disease: A Dynamic Encyclopedia of Disease Mechanisms
Benign tumors of bone include a wide diversity of lesions of various origins, with osseous and cartilaginous lesions being the most common. A number of tumor-like lesions, including secondary aneurysmal bone cysts, unicameral bone cysts, and intraosseous ganglions are considered reactive, that is, nonneoplastic in nature. Some of these entities may have identical histological features and are separated largely on clinical and radiological rounds (i.e., nonossifying fibroma and benign fibrous histiocytoma). Other lesions may have indistinguishable radiological characteristics but occur in different patient populations and exhibit distinct histomorphologic features (i.e., chondroblastoma and giant cell tumor of bone). Thus, correlation of the clinical, radiological, and pathological features of the lesion is required to reach the correct diagnosis for any bone tumor.

View all citing articles on Scopus

^aa: PD Dr. G. Jundt, Bone Tumor Reference Center at the Institute for Pathology, University of Basel, Schönbeinstraße 40, CH-4003 Basel, Switzerland

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Adamantinoma of Long Bones: A Histopathological and Immunohistochemical Study of 23 Cases

Summary

Path Res Pract

Hum Pathol

Path Res Pract

An Analysis of Nine New Cases with Emphasis on Metastasizing Lesions and Fibrous Dysplasia-Like Changes. Hum Pathol

Case report 587: Adamantinoma of the tibia mimicking osteofibrous dysplasia

Skeletal Radiol

Radiologische Befunde beim Adamantinom der langen Röhrenknochen

Fortschr Roentgenstr

Bone and Soft Tissue Tumors

Morphologic Diversity of Long Bone Adamantinoma

The Concept of Differentiated (Regressing) Adamantinoma and Its Relationship to Osteofibrous Dysplasia. Cancer

Bone Tumors

Adamantinoma of the Tibia

An Eccrine Carcinoma. Arch Pathol Lab Med

Staging of Musculoskeletal Neoplasms

Skeletal Radiol

Knochentumoren

Das sogenannte Adamantinom der langen Röhrenknochen

Virchows Archiv Path Anat

Adamantinoma of the Long Bones: Keratin Subclass Immunoreactivity Pattern with Reference to its Histogenesis

Am J Surg Pathol

Bone Tumors