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

Deng, Zhiping; Gong, Lihua; Zhang, Qing; Lin, Hui‐Yi; Ding, Yi; Niu, Xiaohui

doi:10.1186/s13018-020-01769-5

Cited by 10 publications

(7 citation statements)

References 20 publications

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“…In classic adamantinoma, the epithelial component is dominant, with inconspicuous OFD-like regions. In the dedifferentiated adamantinoma, the classic adamantinoma areas gradually transform to a diffuse growing proliferation, where the typical epithelial differentiation is replaced by pleomorphic cells ( 19 , 20 ). The epithelial part of adamantinoma shows co-expression of vimentin, cytokeratin 5, 14 and 19, as well as EMA.…”

Section: Discussionmentioning

confidence: 99%

Rapidly Progressive Classic Adamantinoma of the Spine: Case Report and Literature Review

Lou

Liu

et al. 2022

Front. Oncol.

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Adamantinoma is a locally aggressive or malignant tumor, accounting for <0.5% of all primary bone tumors. The tumor usually progresses slowly, with a relatively promising prognosis. Primary or metastatic spinal adamantinoma of bone are rarer. Only four cases have been documented till date. We present two cases of aggressive spinal adamantinoma whose microphotography and radiographic appearance were unusual, with extensive involvement of multiple segments and rapid progression. Case 1 was a 36-year-old woman, presenting with back pain, progressive numbness and motor weakness, who was diagnosed with metastatic adamantinoma in the T2, T7, L2, and L4. She underwent spondylectomy three times to resect these lesions, respectively. Case 2 was a 68-year-old male with complaints of severe left back pain. MRI revealed destructive changes in T1-T4. He underwent posterior decompression (T1-T3), partial vertebrectomy (T2), fixation and fusion (C5-C7, T4-T6). The pathology of two patients was metastatic spinal adamantinoma, whose primary lesions were from tibia and femoral adamantinoma, respectively. Rapid squamous progression was observed in specimens of T2 and T7 lesions of Case 1 in two months. Twenty-five months after surgery, Case 1 developed paralysis, but she refused to receive further examination and treatment. Two months after surgery, Case 2 presented with an upper back pain again. The MRI revealed an increase in osseous destruction and paravertebral mass size. He was administered radiotherapy, with his upper back pain partially relieved. The biological behavior of classic adamantinoma is highly unpredictable, often exhibiting more aggressive behavior upon recurrence or metastasis. The pathological diagnosis of adamantinoma should be confirmed by preoperative biopsy. En bloc resection with a wide margin is the preferred treatment for primary spinal adamantinoma. Radiation therapy can partially relieve the pain.

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Section: Discussionmentioning

confidence: 99%

Rapidly Progressive Classic Adamantinoma of the Spine: Case Report and Literature Review

Lou

Liu

et al. 2022

Front. Oncol.

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“…better outcome than classic adamantinoma, but long-term follow-up is necessary due to the possibility of local recurrence and late complications (13).…”

Section: Discussionmentioning

confidence: 99%

Osteofibrous dysplasia-like adamantinoma: A case report and literature review

Liu²,

Zhao³

et al. 2022

Front. Oncol.

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backgroundOsteofibrous dysplasia-like adamantinoma (OFD-like adamantinoma), classical adamantinoma and dedifferentiated adamantinoma were previously considered to be three subtypes of adamantinoma of long bones. In the 5th edition of the World Health Organization (WHO) classification of bone tumors in 2020, OFD-like adamantinoma was newly proposed and classified as an intermediate-locally aggressive tumor in other mesenchymal tumors of bone. OFD-like adamantinoma is rare, accounting for only 0.4% of all primary bone tumors. OFD-like adamantinoma is often misdiagnosed due to the insufficient understanding of it. Here we report a case of OFD-like adamantinoma treated in our hospital with a literature review.Case presentationThe patient, a 14-year-old male, had swelling in his right leg with intermittent pain for one year. Plain radiography, computed tomography (CT) and magnetic resonance imaging (MRI) were performed. Based on the radiological and histological examinations, a diagnosis of OFD-like adamantinoma was suspected. After admission, the patient underwent tumor resection of the right tibia, free transplantation of the left fibula and internal fixation. After resection of the tumor, the wound recovered well, the vital signs were stable, and activity was normal. The patient has been followed up for more than a year with no recurrence or distant metastasis.ConclusionOFD-like adamantinoma is a rare primary bone tumor with nonspecific clinical features. Imaging examination can demonstrate the lesion and help diagnosis. The pathological discovery of epithelioid tissue is the key evidence for diagnosis.

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“…Regarding histopathology, adamantinoma is distinguished into osteofibrous-like adamantinoma and classic adamantinoma, although it generally presents many histological types (42). Osteofibrous-like adamantinoma has a better outcome than classic adamantinoma (43). Classic adamantinoma is classified into four patterns of growth namely basaloid, tubular, spindlecell and squamous (22).…”

Section: Histopathological Featuresmentioning

confidence: 99%

“…Compared to classic adamantinoma, which has a dominant epithelial component, osteofibrous-like adamantinoma is composed mainly of osteofibrous tissue. The diagnostic criteria for osteofibrouslike adamantinoma is the predominance of the osteofibrous tissue containing small clusters of epithelial cells and cytokeratin-positive staining (43), while the diagnosis of osteofibrous-like adamantinoma versus osteofibrous dysplasia can only be achieved via detection of a small number of epithelial cells. Adamantinoma exhibits a low mitotic rate.…”

Section: Histopathological Featuresmentioning

confidence: 99%

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Adamantinoma: An Updated Review

Varvarousis

Skandalakis

Barbouti

et al. 2021

In Vivo

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Adamantinoma is a biphasic tumor, with a low potential for malignancy, characterized by clusters of epithelial cells surrounded by a relatively bland spindle-cell osteofibrous component. The aim of the present study was to review the updated data regarding epidemiology; pathogenesis; clinical presentation; radiological, histopathological and ultrastructural findings; and treatment options of adamantinoma. In X-ray, it is usually seen as an eccentric and sometimes central, lobular, lytic lesion with sclerotic margins of overlapping radiolucency, and a characteristic 'soap-bubble' appearance. Magnetic resonance imaging seems to be the most appropriate examination for differential diagnosis between adamantinoma and other skeletal tumors. Histologically, adamantinoma is identified as classic adamantinoma or osteofibrous-like adamantinoma. Classic adamantinoma is classified into four patterns of growth: Basaloid, tubular, spindle cell, and squamous. The preferable treatment of this tumor type is en bloc resection within wide operative margins, which may include suspicious regional lymph nodes, with limb reconstruction and limb salvage.According to the definition of the World Health Organization, adamantinoma is a miscellaneous malignant biphasic tumor and its characteristics are clusters of epithelial cells surrounded by a relatively bland spindle-cell osteofibrous component (1, 2). It is a tumor with low frequency (as it occurs in less than 0.5% of all primary skeletal tumors), in terms of age most often developing between the age of 20 and 50 years, and it is more common in men. It may be located in several bones; however, the mid shaft of the tibia, with or without fibula involvement, is its most common location (3-5). The main symptom reported by patients is pain (6). Treatment of adamantinoma includes extensive resection and reconstruction of bone defect with the use of autograft, allograft, bone transport, and endoprosthesis (7).In our previous article, we presented the epidemiological, clinical, radiological and pathological features as well as treatment options of adamantinoma (4). The aim of the present study was to review the updated data regarding history, incidence, epidemiology, origin and pathogenesis, clinical features, as well as radiological, histopathological and ultrastructural findings, new treatment strategies and prognosis of adamantinoma. The History of AdamantinomaAlthough adamantinoma is an unusual bone tumor, its origin has been debated for almost a century. The term adamantinoma comes from the ancient Greek word adamantinos, which means very hard, as the root word adamas means diamond. The first reported case of a primary skeletal tumor with epithelial characteristics, observed in the ulna, was by Maier (8) in 1900, while in 1913, a physician from Germany, called this tumor 'adamantinoma' as he observed that during embryonic development adamantine 3045 This article is freely accessible online.

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

Cited by 10 publications

References 20 publications

Rapidly Progressive Classic Adamantinoma of the Spine: Case Report and Literature Review

Rapidly Progressive Classic Adamantinoma of the Spine: Case Report and Literature Review

Osteofibrous dysplasia-like adamantinoma: A case report and literature review

Adamantinoma: An Updated Review

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