Key result
Osteofibrous dysplasia-like adamantinoma is linked to better clinical outcomes than classic adamantinoma, including ~0% distant metastasis.
Why the study?
The clinical and molecular characteristics of osteofibrous dysplasia-like adamantinoma differ from classic adamantinoma, but most existing reports are limited to case reports or small series.
Does osteofibrous dysplasia-like adamantinoma have a better clinical outcome compared to classic adamantinoma of long bones?
Cohort (n=23)
No
Does osteofibrous dysplasia-like adamantinoma have a better clinical outcome compared to classic adamantinoma of long bones?
Absolute Event Rate: 14.3% vs 18.8%
Osteofibrous dysplasia-like adamantinoma has a more favorable prognosis with lower metastatic potential compared to classic adamantinoma, supporting extensive curettage when possible.
OFD-like AD may warrant less aggressive surgery than classic AD; extends single-center series but leaves management open pending larger studies.
BACKGROUND: The clinical and molecular characteristics of osteofibrous dysplasia (OFD)-like adamantinoma (AD) differ from those of classic AD. Most reports about OFD-like AD are case reports or small case series. More cases from different centers are still warranted. METHODS: The orthopedic oncology database of our institution was searched to identify patients with AD. The cases of OFD-like and classic AD of the long bones were retrospectively analyzed. Between December 1999 and August 2016, 23 patients were treated for AD, comprising seven with OFD-like AD and 16 with classic AD. The outcomes were compared between AD subtypes. RESULTS: In the OFD-like AD group, four lesions were treated with extensive curettage, while three were treated with wide resection. The median follow-up duration in the OFD-like AD group was 66 months (range 43-131 months). At the end of follow-up, there was only one case of local recurrence (LR) in the OFD-like AD group, giving a LR rate of 14.3% (1/7). No distant metastasis or progression to classic AD was detected in the OFD-like AD group. In the classic AD group, the treatments were below-the-knee amputation in one patient with extensive tibial and fibular lesions, curettage with a bone graft in one patient who was diagnosed with OFD based on a core needle biopsy, hemi-cortical excision and reconstruction in two patients, and segmental resection and reconstruction in 12 patients. At the end of follow-up, there were three cases of LR in the classic AD group, giving a LR rate of 18.8% (3/16); two patients developed lung metastasis after LR and died of the disease at 88 and 126 months after the first surgery in our hospital, respectively. The classic AD group had a metastatic rate of 12.5% (2/16), a final limb salvage rate of 75%, and estimated 5- and 10-year survival rates of 88.9% and 77.1%, respectively. CONCLUSIONS: OFD-like AD has a better outcome than classic AD. For OFD-like AD, extensive curettage is suggested if the tumor extent allows. For classic AD, aggressive resection with wide margins is essential to achieve local control. A long-term follow-up is necessary due to the possibility of late complications.
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Deng et al. (2020) conducted a cohort in Adamantinoma of long bones (n=23). Osteofibrous dysplasia-like adamantinoma vs. Classic adamantinoma was evaluated on Local recurrence rate. Osteofibrous dysplasia-like adamantinoma demonstrated a more favorable outcome than classic adamantinoma, with a 14.3% local recurrence rate and no distant metastasis compared to 18.8% and 12.5%, respectively.
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