Adamantinoma of Bone: A Structured Narrative Review of Clinical Outcomes, Recurrence Patterns, and Metastatic Behaviour
Abstract
Background/Objective: Adamantinoma is a rare, low-grade malignant primary bone tumour with a predilection for the tibial diaphysis. Despite decades of case series and institutional cohorts, the evidence base remains fragmented, and outcomes are inconsistently reported across studies. To synthesise the best available evidence on clinical outcomes, recurrence patterns, metastatic behaviour, and surgical management of skeletal adamantinoma and to appraise the methodological quality of the contributing literature. Methods: A structured narrative review was conducted searching PubMed (294 records) and Web of Science (397 records) from inception to April 2026, yielding approximately 532 unique records after deduplication. Case series of five or more patients with histologically confirmed skeletal adamantinoma were included. A risk-of-bias critique was applied across five domains to each included study. Results: In total, 17 studies, representing more than 900 reported patient entries with possible cohort overlap, formed the primary evidence base. Local recurrence rates for classic adamantinoma (AD) range from 15% to 31%, with metastatic rates from 10% to 27%, predominantly to the lung. The osteofibrous dysplasia-like subtype (OFD-AD) showed no metastases in any series that reports this subtype separately but carries a locally aggressive recurrence rate of 22–43%. Wide resection with uncontaminated margins is the most consistently protective surgical variable (hazard ratio 0.164; p < 0.001). Late recurrences beyond 15 years are documented in multiple series, supporting prolonged surveillance. An MRI-based model for metastatic risk stratification at diagnosis has been proposed but requires external validation. Conclusions: Adamantinoma is more dangerous over a longtime horizon than its low-grade designation implies. Subtype distinction, margin status, and lifelong surveillance are the cornerstones of management. The evidence base carries predominantly moderate to high risk of bias; all conclusions should be interpreted accordingly. A multinational prospective registry remains the most important unmet research need.