Surgical Outcome and Oncological Survival of Osteofibrous Dysplasia-Like and Classic Adamantinomas

Schutgens, Emile; Picci, Piero; Baumhoer, Daniel; Pollock, Robin; Bovée, Judith V.M.G.; Hogendoorn, Pancras C.W.; Dijkstra, P. D. Sander; Rueten‐Budde, Anja J.; Jutte, Paul C.; Traub, Frank; Leithner, Andreas; Tunn, Per‐Ulf; Funovics, Philipp; Sys, Gwen; San-Julián, Mikel; Schaap, Gerard R.; Dürr, Hans Roland; Hardes, Jendrik; Healey, John H.; Capanna, Rodolfo; Biau, David; Gomez‐Brouchet, Anne; Wunder, Jay S.; Cosker, Thomas; Laitinen, Minna; Niu, Xiaohui; Kostiuk, V.; Sande, Michiel A. J. van de

doi:10.2106/jbjs.19.01056

Cited by 14 publications

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“…However, the biological behavior of adamantinoma is highly unpredictable, and usually presents with a more aggressive pattern when it relapses ( 25 , 26 ). In the cohort reported by Schutgens EM et al., 27% (6/28) of patients with recurrent adamantinoma developed local recurrence after more than ten years of follow-up ( 17 ). Adamantinoma will metastasize as well, mainly to the lungs and local lymph nodes.…”

Section: Discussionmentioning

confidence: 99%

“…Clinical symptoms or radiographic abnormalities can last for more than 30 years before presentation to the hospital ( 2 ). Clinically, pathological fracture occurs in 16%-23% of patients ( 17 ). In our two cases and previous reports of spinal adamantinoma, the patients often presented with back pain and neurological dysfunction ( 8 , 9 , 11 ).…”

Section: Discussionmentioning

confidence: 99%

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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%

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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“…‘En bloc resection with wide margin of the OFD/AD, and surveillance of the OFD’ [ 11–13 ] considering OFD/AD as a histological subtype of AD [ 4 , 12 , 13 ]. A recent multicenter study proposed to reclassify OFD/AD as an aggressive local intermediate and not as a variant of adamantinoma [ 11 ]. The authors based their study on the results of surgical treatment of 128 OFD/AD and 190 AD.…”

Section: Discussionmentioning

confidence: 99%

“…deserves surgical management, even in the case of OFD [ 6 ]. Second, the lesion only affected the isolated fibula, which does not require any bone reconstruction in a child, compared with the classical tibial lesions of the OFD-AD spectrum [ 1 , 2 , 7 , 11–13 ]. However, in view of the high risk of recurrence reported by Schutgens et al.…”

Section: Discussionmentioning

confidence: 99%

Osteofibrous dysplasia-like adamantinoma of isolated fibula in a child mimicking chronic osteomyelitis with pathological fracture

Goetz

Dumitriu

Galant

et al. 2022

Journal of Surgical Case Reports

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The occurrence of a pathological fracture in children requires a rigorous diagnostic approach in order to establish the etiology and to develop a precise therapeutic strategy. Several causes are associated with these fractures, the most frequent being benign tumors in children in developed countries and chronic osteomyelitis in developing countries. More rarely, malignant tumors must however always be considered. The differential diagnosis on imaging may be difficult to establish between bone tumors and chronic infection. Surgical biopsy is therefore often performed to establish the precise origin of the fracture. We report the case of an adamantinoma (osteofibrous dysplasia-like) of the fibula in a 7-year-old child, discovered during the management of a pathologic fracture. The presumed diagnosis before biopsy was chronic osteomyelitis. A 14-cm-resection of the affected fibula was performed with good functional result. Differential diagnosis between adamantinoma, osteofibrous dysplasia and osteofibrous dysplasia-like adamantinoma remains very challenging.

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“…Thirty percent of patients may live with the above symptoms for many years, 5 years or more, without proceeding to a medical examination. This tumor causes pathological fracture in 16-23% of patients (34), while the presence of trauma has been reported in about 25% to 60% of cases (5,34). Adamantinoma is a malignant tumor and can metastasize, mainly to the lungs and adjacent lymph nodes.…”

Section: Clinical Featuresmentioning

confidence: 99%

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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Surgical Outcome and Oncological Survival of Osteofibrous Dysplasia-Like and Classic Adamantinomas

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Cited by 14 publications

References 25 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 of isolated fibula in a child mimicking chronic osteomyelitis with pathological fracture

Adamantinoma: An Updated Review

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