First Confirmed Metastatic Adamantinoma of the Spine: Case Report and Literature Review

Abstract

Study Design Case report and literature review. Objective To present the first case of metastatic adamantinoma of the spine with immunohistochemical confirmation and an updated literature review. Summary of Background Data Spinal metastatic disease could be difficult to diagnose because of the multiple differential diagnoses involved. Spinal surgeons must be aware of unusual primary extra-axial tumors that metastasize to the spine because in certain cases the primary surgery must determine the prognosis of this lesion. Methods We present a fully documented case of a middle-aged man with tibial adamantinoma who developed spine metastasis, confirmed by immunohistochemistry. A literature review was done. Results Based on clinical, imaging, and pathology findings, we provide evidence for the first proven metastatic adamantinoma of the spine, adding this condition to the long list of differential diagnoses of secondary spinal disease. Conclusions Adamantinoma is a very rare bone tumor representing ∼1% of primary bone tumors. Spinal metastatic adamantinoma with immunohistochemical confirmation has not been described previously. Due to the lack of specific image findings or serum markers and multiple differential diagnoses, biopsy with immunohistochemical confirmation is mandatory, because “en block resection” might prove to be curative treatment.

Recurrent adamantinoma metastatic to the spine: Clinical and imaging considerations

“…A locally aggressive tumor, adamantinomas have been reported to metastasize in approximately 10% to 20% of cases with the most common sites being the lungs and less likely lymph nodes. Metastasis to a secondary skeletal site is exceedingly rare, particularly to the spine where there is only one prior report (reference) [2 , 4] . Primary adamantinoma of the spine is also rare, with six cases described in the literature.…”

“…The differential diagnosis of adamtinomatous lesions, in general, includes chondromyxoid fibroma, fibrous dysplasia, osteofibrous dysplasia, Ewing sarcoma, and osteosarcoma [2 , 7 , 8] . Radiographic and CT imaging characteristics of adamantinoma include a multilocular or slightly expansile and lytic, cortically based lesion with scattered areas of lysis interspersed with sclerosis and a relative lack of periosteal reaction [9 , 10 , 11] .…”

“…The probability of local recurrence of this lesion is directly dependent upon the success of index surgical resection, ideally including clear margins. Adamantinoma is highly resistant to both chemotherapy and radiotherapy [2 , 14] . However, there is an overall insufficient amount of data to comment on the optimal treatment of metastatic adamantinomatous lesions involving the spine [15] .…”

“…A low-grade malignancy, adamantinoma has rarely been identified to metastasize distantly, primarily to the lung, osseous structures, lymph nodes, pericardium, and liver. Metastatic involvement of the spine is an extremely rare entity with only 1 case documented in the literature to date [2] . To put this into perspective, spinal metastases occur in approximately 40% of patients who succumb to cancer, and at least 5-10% of all patients suffering from cancer will present with symptomatic spinal metastases at some point during their disease course [3] .…”

A case of progressive, metastatic adamantinoma in a pediatric patient

“…4,[6][7][8][9][10] There have been a few case reports of bone metastases, but no previous reports of widespread bony metastatic disease as in our patient's case. [11][12][13] The presence of widespread metastases and rapid progression was a unique feature of this case. Her presentation was likely due to significant delays in care, initial misdiagnosis, and treatment with bone curettage and grafting, which can promote hematogenous spread.…”