Adamantinomatous tumors: Long‐term follow‐up study of 20 patients treated at a single institution

Abstract

Background and Objectives Adamantinomas are primary, low‐grade malignant tumors of the bone that have metastatic potential to the lungs, lymph nodes, and other regions. The rarity of this disease and its nonspecific symptoms complicate diagnosis. Materials and Methods Records for 20 patients who underwent treatment for adamantinoma from 1975 to 2018 were reviewed for demographic, clinical, and pathological data, treatment details, postoperative complications, and outcomes. Results Patients presented at a median age of 22 years (1‐79 years): 14 patients had a localized primary tumor, three presented with local recurrence, and three with metastatic disease. Median tumor size was 5.7 cm (0.5‐15.5 cm). Wide excision was performed primarily in 15 cases; the remaining five patients underwent intralesional curettage. At a median follow‐up of 7.3 years, 14 patients had no evidence of disease; two patients were alive with disease, and four patients died from the disease. Local recurrence and distant metastasis occurred at a median of 11.4 years (6 month‐19 years) and 15.8 years (4 month‐23 years) after diagnosis. Conclusions Adequate histopathological diagnosis is crucial to avoid misdiagnosis of this rare tumor. Local and distant recuAbs_Para_meprrence can occur more than 20 years after the initial diagnosis. Life‐long follow‐up with clinical examination and imaging is required.

Rapidly Progressive Classic Adamantinoma of the Spine: Case Report and Literature Review

“…In a study of 70 patients with adamantinoma, the 5-year survival rate was at 95%-98.8% ( 12 , 22 ), while the 10-year survival rate was 87.2% ( 24 ). However, the biological behavior of adamantinoma is highly unpredictable, and usually presents with a more aggressive pattern when it relapses ( 25 , 26 ). In the cohort reported by Schutgens EM et al., 27% (6/28) of patients with recurrent adamantinoma developed local recurrence after more than ten years of follow-up ( 17 ).…”

Adamantinoma: An Updated Review

“…As already mentioned, adamantinoma is a biphasic tumor, usually consisting of epithelial cells that stain positively for keratins 14 and 19 inside spindle-celled fibrous tissue (20); it is then a tumor of epithelial origin. In recent years, several hypotheses have been made regarding the origin of this tumor type.…”

“…Regarding gender, it is found more often in men, with a ratio of five to four (3,4,6,16). Commonly affected patients are those between the age of 20 to 50 years; although adamantinoma presents earlier in females, more specifically between 11 and 30 years old, while in males between 20 to 50 years, although the range of ages is from 3 to 86 years (19,20). However, the tumor is rare in childhood (21).…”

“…The main histological patterns of classic adamantinoma include basaloid, tubular, spindle-cell and squamous patterns. On the other hand, differentiated adamantinoma presents as osteofibrous dysplasia-like, regressive, juvenile and intracortical (20).…”

“…In a review of 70 patients, the 10-year survival rate was reported to be as high as 87.2% (59), while the 5-year survival rate was higher at 95-98.8% (6,23). The biological behavior of an adamantinoma is highly unpredictable (30) and generally recurrence presents with a more progressive behavior (20). About 15% of the patients develop metastases to the lungs and lymph nodes, although rarely is the skeleton, liver and brain affected (1,60,61).…”