Adamantinoma of the distal femur diagnosed 5 years after initial surgery: a case report

Abstract

BackgroundAdamantinoma arising in the femur is extremely rare. We report a case of an adamantinoma occurring in the right medial femoral condyle that was diagnosed 5 years after the primary surgery.Case presentationA 74-year-old Asian woman first complained of right knee pain without any cause. Radiographs demonstrated a 4×4.5 cm osteolytic lesion in her medial femoral condyle. Magnetic resonance imaging revealed a lesion which showed low signal on both T1 and T2-weighted image, and enhanced signal with gadolinium contrast administration. She underwent a wide resection of the lesion and was reconstructed with a tumor endoprosthesis. On histological examination, the tumor showed clusters of spindle-shaped and squamoid epithelial cells among the fibrous stroma. Adamantinoma was considered, however, the diagnosis was inconclusive due to the unusual localization and her age. Moreover, it was difficult to exclude metastatic carcinoma. Five years later, she was diagnosed with an abnormal shadow occupying the upper lobe of her right lung in a routine physical examination. She subsequently underwent a resection of the lung mass which histologically showed proliferation of spindle-shaped and squamoid epithelial cells. The histological similarity of the lung tumor and the femoral tumor led to the diagnosis of adamantinoma arising in her right medial femoral condyle with metastasis to the upper lobe of her right lung.ConclusionIn this case report, we report the clinical, radiographic, and histological features of an adamantinoma arising in the distal femur with a review of the literature.

Epidemiological Study of Adamantinoma from US Surveillance, Epidemiology, and End Results Program: III Retrospective Analysis

“…Adamantinoma is most commonly located in long tubular bones such as the tibia fibula and ulna and is rarely located in the femur humerus and radius [20][21][22]. In our data, at least 96.7% of the lesions were located in the long bones of the lower extremities.…”

17 Yaşında Erkek Hastada Sol Proksimal Tibiada Ağrısız Lezyon: Adamantinoma

“…Adamantinoma olgularının çoğunluğu uzun kemiklerin merkezinde görülür, bu lezyonların %85'i tibiada görülür. Daha nadir olarak fibula ve ulna da etkilenir (5,9). Bizim olgumuz da literatüre uygun şekilde tibia yerleşimli idi.…”

“…Kemoterapi veya radyoterapinin tedavide yeri yoktur. Lokal rekürrens ve uzak metastaz nadirdir, metastaz gelişirse en sık akciğerlere gelişir (7,9,10).…”

“…Adamantinoma tedavisi lezyonun geniş rezeksiyonu ve kemik defektinin rekonstrüksiyonudur (9,10).Kemoterapi veya radyoterapinin tedavide yeri yoktur. Lokal rekürrens ve uzak metastaz nadirdir, metastaz gelişirse en sık akciğerlere gelişir (7,9,10).Biz bu çalışmada sağ tibia proksimali yerleşimli adamantinoması olan bir hastanın presentasyon, görüntüleme bulguları, tedavisi ve takibini tanımladık.…”

“…Bir malign kemik tümörü olan adamantinomanın tedavisi hakkında ise, nadir görülmesi nedeniyle literatür verileri sınırlıdır. Adamantinoma tanılı tüm olgularda tercih edilen tedavi tümörün geniş rezeksiyonu ve defektif alanın rekonstrüksiyondur (1,5,9,16,17).…”