Adamantinoma of the long bones
Case report and bibliographic review

CH.K. KYRIAKOPOULOS, A.F. MAVROGENIS, G. NOMIKOS, E. FANDRIDIS, D. KOULALIS,
P.I. PAPANGELOPOULOS
First Department of Orthopaedics, Athens University

ABSTRACT
Adamantinoma of the long bones is a rare, primary, low-grade, slow growing malignant bone tumor, composed of epithelial cells within a fibrous or fibrous-bone mesenchymal reactive stroma. In most of the cases, the tumor is usually located in the anterior cortex of the tibial shaft. The tumor usually spreads to the lungs, the regional lymph nodes, or the bones.
The wide tumor excision and limb salvage reconstruction surgery, or an amputation, are the current surgical treatment options. Chemotherapy and adjuvant radiotherapy have not been shown to be effective.

Key words: adamantinoma, ameloblastoma, wide excision, limb salvage, amputation.

CASE REPORT
A 23-year-old man observed the emergence of a gradually increasing mass on the anterior surface of the middle third of his right tibia. The bulge emerged almost three weeks after a tibial injury, and was painful during the leg movements.
The clinical examination of the patient, revealed a sensitive during palpation mass at the middle part of the right tibia. The supernatant skin was normal. No swollen regional popliteal or inguinal lymph nodes were present. The adjacent joints of the knee and ankle were normal. The family and personal history of the patient was negative.
Simple X-rays of the right tibia, revealed a sizeable, multifocal osteolytic lesion that was located in the inner-anterior cortex of the medial part of the right tibial shaft, with concomitant local periosteal reaction (figure 1). Bone scanning (99mTC-MDP) revealed a local increase in the radioisotope uptake at the area corresponding to the radiographic findings. MRI revealed a sizeable, heterogeneous mass with irregular borders, the dimensions of which were 1.5 x 3 x 8cm, located at the middle part of the right tibial shaft (figure 2). The lesion presented intramedullar expansion and was spreading to the external margin of the tibial cortex. No soft tissue mass was evident.
An open biopsy was performed and tissue specimens were taken. The diagnosis of adamantinoma was given by the histologic examination.
The patient underwent a wide tumor excision and limb salvage surgical operation. The surgical borders were negative for malignancy. The tibial restoration was performed by the implantation of a cadaveric shaft allograft of 15cm of length. The stabilization of the allograft was succeeded by the use of bone cement with vancomycin and a dynamic compression plaque (LCDCP, Synthes) with sixteen holes. Moreover, an autograft taken from the ilium was placed at the junction site with the bone allograft.
The histologic evaluation of the specimen revealed a mass; its dimensions were 1.5 x 3 x 8cm. The mass had eroded the anterior tibial cortex. The excision borders of the tumor (>3.5cm) were negative for malignancy. There were islets of epithelial cells within a stroma of fibrous connective tissue (figure 3).
Postoperatively, we placed a femoral-tibial-ankle cast for 6 weeks and then a splint of plaster for another 6 weeks. The radiological evaluation, after this period, revealed porosis of the allograft at the proximal tibial part, something that was not evident at the distal one. Because of the lack of porosis of the allograft at the distal part of the tibia, a second surgical operation was performed, during which we placed a bone autograft from the crista iliaca (figure 4) and a cast for 4 more weeks. After this, the porosis of the allograft was successful.
During the last follow-up examination, height years after the diagnosis was established, there was full radiographic incorporation of the allograft, while the patient was healthy and free of local recurrence, with a slight limitation of his leg function and activities.

1.
Figure 1. Anteroposterior (a) and lateral (B) X-ray that shows a multifocal lesion in the size of 9.5cm in the anterior cortex of the right tibial shaft.

2.
Figure 2. MRI reveals a sizeable mass with irregular borders at the middle of the right tibial shaft. The lesion is situated in the external-anterior cortex of the bone and expands into the medullary cavity. Periosteal reaction is visible at the external border of the anterior cortex. No soft tissue mass is noticed.

3.
Figure 3. A typical histological picture of an adamantinoma. (A) At low magnification, islets of epithelial cells which are surrounded by fibrous connective tissue are visible. (B) At high magnification, loose piles of epithelial cells within a fibrous stroma are present.

4.
Figure 4. Anteroposterior (A) and lateral (B) X-rays after a wide surgical excision of the tumor and a limb salvage surgical operation performed by the use of a diaphysic allograft and an iliac autograft. Incorporation of the allograft without any radiographic signs of local recurrence is present.

DISCUSSION
Adamantinoma of the long bones is an uncommon, primary, low grade, slow growing malignant bone tumor. Adamantinoma presents a wide variety of histologic types, with malignant biologic behavior[10,13,21,24,28,44,50,52,54,60].
Generally it is comprised by epithelial cells within a stroma of fibrous or bone-fibrous connective tissue[15,19,21,22,33,34,36,37,44,45,52,61]. Adamantinoma has the same histological picture as the amelovlastoma or adamantinovlastoma of the mandible, but without any other clinical relation with these two entities.
Adamantinoma of the long bones constitutes about 0.3 to 1% of all malignant bone tumors[17]. Adamantinomas are usually located at the diaphysis and less at the metaphysis of the long bones. The anterior cortex of the tibia is the more common (90%) site of emergence for this tumor[17,27,43]. Fibula insult is much less common, and it usually coexists with the insult of the ipsilateral tibia. There are reports of the tumor at the olecranon[56], the ribs, the radius, the spinal column, the metatarsal bones and the humerus[4,5,12,46,62].
Adamantinoma usually affects patients after their skeletal maturation, between the second and fifth decade. The incidence of the tumor among men and women is 1,3-2:110,13,17,21,24,28,44,50,60. Adamantinomas have been reported in children, but their histological picture differs from that of the adults, and resembles to the histological picture of fibrous-bone dysplasia. The most common adamantinoma type in children is called "differentiated adamantinoma" and it follows a relatively benign biological course, with a better prognosis and outcome[35].
The typical clinical features are a gradually growing mass at the tibia, and a dull and intermittent pain, which never becomes intense and persistent. Pathological fracture of the affected bone is possible. A history of previous injury, trauma or fracture, several months or years before the first symptoms is reported in about 60% of the patients. Advanced or recurrent lesions are usually related to infiltration of the surrounding soft tissue[17,61]. Adamantinomas usually spreads to the lungs, the regional lymph nodes or the bones[56,23]. In rare cases, lung metastasis are present during diagnosis[2,16,17,48]. Furthermore, the emergence of hypercalcemia and shock, because of paraneoplastic syndrome and pancreatitis has been reported[2,63].
In simple X-rays, adamantinoma presents a central lytic core, surrounded by multiple, marked radiolucent zones of varying width and a sclerosing margin, which causes the bulging of the anterior part of the cortex. The radiographic features of the adamantinoma are similar to those of the fibrous-bone dysplasia. The differential diagnosis between these two entities should be clinical (fibrosseous dysplasia causes no pain, whereas pain is a common symptom in patients with adamantinoma), while it is also related to the physical course of the disease (the fibroid processes of the bones are suspended after the maturation of the bone, while adamantinomas keep growing after the adulthood)[7,17,18].
Bone scanning presents an increased uptake of the radioisotope at the site where the lesion is radiographically located[7,17,61].
Computed tomography reveals the osteolytic lesion eroding the cortex and usually expanding to the surrounding soft tissue[17,18].
Magnetic resonance imaging is useful for the differential diagnosis of adamantinoma from other bone lesions, like fibrous-bone dysplasia, osteosarcoma and Ewing sarcoma. Moreover, it gives information about the exact location and extent of the tumor inside the bone and the infiltration of the soft tissue[7,30]. In T1 weighted imaging the lesion presents a low intensity signal, while in T2 weighted imaging it presents a high intensity signal, that doesn't decline by the use of fat suppression technique[61].
Adamantinomas present a wide variety of histologic types, imitating many primary or metastatic bone tumors[17,29]. In histological preparations, two cell components (epithelial and mesenchymal) are discernible, but it is not feasible to distinguish the epithelial or double origin of the tumor. The presence of the epithelial cell component was proven in the past, by electron microscope studies and immunohistochemical assays, where the expression of cytokeratines of the basal cells was confirmed on the tumor cell surface[21,22,50,52]. Four epithelial histologic types have been described: the basal-carcinoid type, with cells in strings and islets similar to those of the basal cell carcinoma, the fusocellular type, similar to the previous one, but without any peripheral matrix of "palisade" cells, the solenoid type, which is composed by small branched tubules and faveolar cavities, that have one or more layers of cuboid, cylindrical cells, and the squamiform type, composed by squamus-cell nodules. The presence of two or more histologic types in the same tumor is unusual[9,17,24]. The second histologic component is the mesenchymal (fibroid) cellular stroma. The mesenchymal component consists of immature fusiform cells which are arranged between collagen bundles. The size and shape of the cells varies, while the asteroid shape of the nuclei resembles to that of the primitive dysplastic mesenchymal cells that compose the stroma of the fibrous-bone dysplasia[61].
Recent studies with flow cytometry and DNA imaging, immunohistochemical detection of p53 gene, detection of the heterozygous state of the p53 gene with polymerase chain reaction (PCR), and immunohistochemical control studies of the expression of the type 2 fibroblast growth factor (FGF-2), the epidermal growth factor (EGF) and their corresponding receptors, FGFR-1 and EGFR, as also the Ki-67 marker have revealed that most likely the malignant component of adamantinoma are the cells of epithelial phenotype[6,7,21,22,31,32].
The histologic differential diagnosis of adamantinoma should include the metastatic epithelial tumors of the bones, the hemangioepithelioma, hemangiosarcoma, fibrous and fibrous-bone dysplasia, and the non-osteogenetic and chondromyxoid fibroma. The "differentiated adamantinoma", which is diagnosed during childhood presents similar histologic features to the fibrous-bone dysplasia and seems to constitute the precursor lesion of the classic adamantinoma[13,53]. However, the relation between this particular histologic type of adamantinoma and the fibrous-bone dysplasia is still obscure[3,8,24,25,49,68]. The accurate diagnosis is attained by fine needle aspiration biopsy[1,17,41].
The biological behavior of the adamantinoma cannot be accurately determined, because of the sparseness of the tumor, while facts about the safer and more effective treatment type are not yet sufficient. Moreover, besides the fact that there is not much experience, chemotherapy and adjuvant radiotherapy of the adamantinoma seem to be ineffective because of the low grade and high radioresistance of the tumor[24,38,67].
Treatment of choice for the adamantinoma is the wide (en block) surgical excision of the tumor with a limb salvage reconstruction surgical operation or a limb amputation[9,17,19,21,24,28,33,44,50,51,60,61]. The presence of metastasis later on is not uncommon, especially in patients that underwent a defective surgical excision[14,17,33] and may require an amputation in the future[50].
The limb salvage and reconstruction surgical operation may be achieved with the use of a diaphysic or cadaveric bone-cartilage allograft, or an autograft of a vascularized or not fibula, a metal prosthesis and straining/stretching osteogenesis[11,17,19,39,40,42,47,50,55,57,61,65,66].
The almost benign biology of the tumor and its slow growing results in a long latent period between the emergence of the symptoms and the diagnosis, and is connected to the high survival rates even after a local recurrence or after the presence of regional lymph node or even distal pneumonic metastasis[17,19,29].
The local recurrence rates are reported to be 18,6% in patients that underwent a wide tumor surgical excision. The local recurrence usually emerges five to ten years after the diagnosis and surgical treatment of the adamantinoma. Very late local recurrences, twenty four and thirty six years after the diagnosis have also been reported[17,58]. However, no relation between the potential of local recurrence and the stage of the disease, the duration of symptoms, the age of the patient and the type of the graft has been established[35,50]. High recurrence rates have been observed in patients whom the surgical excision was not wide[24,50,56]. The wide surgical excision is connected with lower local recurrence rates compared to the excision through the tumor and the marginal excision. Moreover, the wide excision is recommended not only for the removal of the primary tumor, but also of the local recurrences[48].
The adamantinoma prognosis depends on the type of the treatment. Wide surgical excision of the tumor is related to the most favorable prognosis. Adamantinoma usually appears as an endosteal intracortical sarcoma of stage I-A, but with a potential of developing into metastatic disease (stage III), from which about 15% of the patients will die. Young women seem to be in a higher risk of early metastasis, with a mean age of death of thirty three years, compared to forty height years in men[61]. Surgical removal of the pneumonic metastasis usually has good results. In a patient series, the mortality rates were 13% to 18% and the metastasis rates 10% to 30%[21,24,33,44].
In conclusion, adamantinoma of the long bones is a rare, primary, slow-growing, low grade malignant bone tumor. It is expressed in a wide variety of histologic types and composed by an epithelial cell component with signs of malignancy and a fibroid mesenchymal reactive component. Most of the tumors are located in the tibial shaft. The tumor usually metastasizes to the lungs, the regional lymph nodes and the bones. Treatment of choice for the adamantinoma is the wide surgical excision with a limb salvage operation or an amputation.

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