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Adamantinoma

DEFINITION AND PATHOGENESIS

 

IMPORTANCE

  • Must be distinguished from osteofibrous dysplasia (but related to it) and metastatic carcinoma
  • Variability in diagnositc criteria between osteofibrous dysplasia, osteofibrous dysplasia-like adamantinoma and classic adamantinoma has posed obstacles in defining the biology and management of these lesions

CLINICAL FEATURES

  • 87% occur in tibia (most common) and fibula, other bones include ulna, femur, humerus, small bones of hands and feet
  • Male:female = 1.3-1.7:1
  • Mean age 30-31
  • Average age: female 27, male 44 (35 overall)
    • Youngest age reported: 3 yrs
      • Abundant epithelial component with formation of keratin pearls reported in a pt aged 3 yrs
  • Dull aching pain and gradual swelling
  • Ulceration has been reported
  • Up to 13% with pathological fx
  • Unusual locations
  • Unusual presentation

RADIOLOGIC FEATURES

  • Diaphyseal to metadiaphyseal, usually 6-7cm in length and up to a mean of 13.2 cm
    • 7/8 with moth-eaten marginsin adamantinoma
      • (5/11 with moth-eaten margins in osteofibrous dysplasia-like adamantinoma)
      • (2/5 with osteofibrous dysplasia)
  • Central or eccentric elongated, well circumscribed, honeycomb or bubbly focus of osteolysis ± sclerosis
  • Advanced lesions may have a soft tissue mass (also recurrences)
  • Purely intracortical lesions do occur as do single cystic lesions
  • Metastases at time of dx reported 3%
  • Bone scan
  • MRI
    • Provides the most accurate information about extent and invasiveness
    • Anterior cortical involvement (19/22)
    • Extension into the bone marrow (12/22)
    • Two morphologic patterns
      • Solitary lobulated focus
      • Pattern of multiple small nodules in one or more foci
        • High signel intensity foci on T2- and T1-weighted contrast-enhanced images interspersed with normal appearing cortical or spongious bone (6/22)
    • Intense and homogeneous static enhancement

GROSS PATHOLOGY

  • Gray white and firm, cystic degeneration, foci of hemorrhage, may extend to dermis

HISTOLOGIC/MOLECULAR FEATURES

DIFFERENTIAL CLINICOPATHOLOGIC DIAGNOSIS

  • Metastatic carcinoma
  • Angiosarcoma and myxoid angioblastoma (factor VIII +)
  • Fibrous dysplasia
  • Osteosarcoma with fibroblastic differentiation
  • Osteofibrous dysplasia (? precursor to adamantinoma)
  • Adamantinoma-like Ewing sarcoma
    • See below under SPECIAL CONSIDERATIONS
  • Rarely
    • Intraosseous glomus tumor of the fibula reported mimicking adamantinoma radiographically

DISEASE COURSE AND TREATMENT

  • Tx is wide en blocwide resection
  • Average time to LR is 5.7 years
    • LR reported in up to 31%
    • LR develops if management is intralesional or marginal in nature or if periosteum is not resected
      • LR and lung metastases 19 yrs after curettage for what was diagnosed as "fibrous dysplasia"
      • LR 20 yrs after resection reported
      • LR 24 yrsafter dx and resection associated with lung metastases
        • LR and metastases surgically removed and short term NED x 3 yrs reported
    • Treatment of LR with wide resection has been reported successful in selected cases
  • Metastases to lungs, bones, lymph nodes (7%), pericardium, liver, retroperitoneum, and posterior cul-de-sac** 10-30% reported (higher mitotic rate with higher risk)
    • Dx of pulmonary metastases has been reported by pulmonary brushing cytology
    • Reported 16 yrs after knee disarticulation with metastasis to contralateral leg and lungs
    • Reported to lung after 6 yrs after a BKA
    • A series of osseous metastases has been reported
      • A report of skeletal metastasis 4 yrs after resection reported
      • Reported in ipsilateral femur 5 yrs after AKA
    • Skeletal and pulmonary metastases have been reported after intralesional curettage and IM nailing of a tibia six years after surgery without reported evidence of LR
  • Mean survival 12.8 yrs in metastatic disease
  • Vascularized osteoseptocutaneous free flap has been used to reconstruct ulnar lesions
  • 10 yr survival 87.2%
  • Chemotherapy
  • XRT
    • Hypercalcemiawith a parathormone-like substance released by metastatic adamantinoma to the lung rapidly responded with normalization of serum calcium levels
      • Gradual return of elevated calcium levels occured after 15 months

SPECIAL CONSIDERATIONS

    • Similar features
      • Similar histologic features
      • Similar immunohistochemistry
      • Shared clonal abnormalities
        • Chromosomes 7, 8, 12, and 21 in both
        • (Chromosome 19 only in adamantinoma, not in osteofibrous dysplasia)
      • Overlapping skeletal distribution
      • Simultaneous occurrence in tibia and fibula
      • Simultaneous occurrence in the ulna
    • 3/16 ptstreated for what was felt to be osteofibrous dysplasia were confirmed to have adamantinoma on final histological examination
      • Because of LR with curettage of osteofibrous dysplasia (6/16) and the risk of adamantinoma (3/16), extraperiosteal excision of osteofibrous dysplasia has been advocated
      • Strict surveillancehas been reported as mandatory in osteofibrous dysplasia
        • Systematic biopsy in case of onset of pain or increased tumor volume in pts with osteofibrous dysplasia has been recommended
    • Presence of keratin-positive epithelial cells within the stroma of osteofibrous dysplasia must notbe present to confirm progression to adamantinoma
      • Cases have been reported where osteofibrous dysplasia-like adamantinoma have been misdiagnosed as osteofibrous dysplasia
    • Common expression of oncoproteins and bone matrix proteins:

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  • These investigators from Boston studied three cases of adamantinoma with electron microscopy and immunohistochemistry. Well-differentiated epithelial cells and glandular structures were present in the tubular pattern while in the basaloid pattern, less differentiated epithelial cells with discohesion were seen in the central epithelial masses. This study established the epithelial nature of some tubular structures with slit-like lumina, easily misinterpreted as capillaries by light microscopy. Results also showed that the irregular spaces observed within the basaloid pattern probably result from cell discohesion. Moreover, this investigation demonstrates the epithelial nature of a subset of spindle cells within the stroma of adamantinoma and offers ultrastructural evidence for a probable mesenchymal-epithelial transformation as its histogenesis.


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