Background: Adamantinoma of long bones is
a rare tumor. Published reviews of the orthopaedic management of adamantinoma
have involved limited follow-up of small numbers of patients. The
oncological aggressiveness of this tumor is unknown. Limb salvage
is currently the treatment of choice for most adamantinomas. The
purpose of this study was to evaluate the characteristics of adamantinoma
of long bones as well as the oncological outcome and the complications
of limb salvage operations.
Methods: A retrospective study was designed
to evaluate the clinical outcomes of limb salvage operations for the
treatment of adamantinoma. Data on seventy biopsy-proven cases of
adamantinoma treated between 1982 and 1992 at twenty-three different cancer
centers in Europe and North America were obtained.
Results: The median duration of follow-up was
7.0 years. The male:female ratio was 3:2, and the mean age was thirty-one
years. Limb salvage was attempted in 91 percent (sixty-four) of
the seventy patients, and the final rate of limb preservation was
84 percent (fifty-nine of seventy). Wide operative margins were
obtained in 92 percent (fifty-eight) of sixty-three patients. An
intercalary allograft was used to reconstruct the segmental bone
defect in 51 percent (thirty-six) of the seventy patients. Reconstruction-related
complications occurred in 48 percent (thirty) of sixty-two patients.
Nonunion and fracture were the most common complications, occurring
in 24 percent (fifteen) and 23 percent (fourteen) of sixty-two patients,
respectively. Kaplan-Meier analysis demonstrated a rate of local recurrence
of 18.6 percent at ten years. Wide operative margins were associated
with a lower rate of local recurrence than marginal or intralesional margins
were (p < 0.00005). Kaplan-Meier analysis showed a survival
rate of 87.2 percent at ten years. There were no significant relationships
between survival and the stage of the tumor (p = 0.058), duration
of symptoms (p = 0.90), gender (p = 0.79), or wide operative margins
(p = 0.14).
Conclusions: Current treatment of adamantinoma,
including en bloc tumor resection with wide operative margins and
limb salvage, provides lower rates of local recurrence than has
been previously reported. In the present study, the limb preservation
rate was 84 percent (fifty-nine of seventy), and the survival rate
was 87.2 percent at ten years. The rate of complications related
to the limb reconstruction was high.