DEFINITION AND PATHOGENESIS
- Benign lesion with cyst-like walls filled with blood
- Vascular anomaly may be begun by trauma or tumor
- Can be mistaken for a malignancy due to fast rate and destructive nature of early growth as well as exuberant cellularity
- May be associated with other lesions, particularly
- Fibrous dysplasia
- Most occur prior to age 20, rarely after age 50
- Mean age for pelvic lesions 15.5 (2-44)
- Symptoms may be of short duration, often <3mos, with pain ? swelling
- Average duration of sxs for pelvic lesions 7 months
- 95% of pelvic lesions present with pain (thigh, groin, LBP, buttocks, leg), 40% with ? hip ROM, 10% with mass (Ö rectal exam), neurologic signs/sxs (67% of sacral lesions) including urinary retention, ? anal tone
- M:F = 1:1.16 from compiled data
- Neurological sxs
- Peroneal nerve palsy from proximal fibular location reported
- Spinal lesions may present with neurological sxs/signs
- Hemothorax froma a T4 ABC has been reported
- Has been reported after fx or trauma (up to 23%)
- Has been reported causing proptosis and epiphora in an ethmoid location
- Labyrinthine fistula reported from ABC within fibrous dysplasia
- Expansile, often involving long bones, pelvis, posterior spinal elements, and may become massive in size
- Extension across the physis is rarely reported
- Deformity and limb length discrepancy reported
- ?40-44% of spinal lesions involve multiple vertebrae
- Anterior extension and "vertebral plana" may occur
- Three (not always distinct) phases:
- Early, incipient, or initial phase: lytic area may be small or large, geographic or poorly circumscribed
- Midphase or proliferation stage: extreme cortical destruction, "blowout", Codman's triangles, very worrisome aggressive features, including an incomplete rim of periosteal bone, and the "finger-in-the-balloon" sign
- Late, equilibrium, or inactivation phase: bone's reaction to the lesion has caught up, with rim of periosteum
- Campanacci radiographic classification system
- Type 1: the cyst is within the center of the bone with little or no cortical expansion
- Type 2: the cyst is within the entirety of the involved segment of bone with severe cortical expansion
- Type 3: the cyst is eccentrically located within the bone with little or no cortical expansion
- Type 4: the cyst is supbperiosteal with erosion of the cortex (least common)
- Type 5: the cyst expands into the soft tissues with periosteal elevation, cortical destruction, and cancel-lous osteolysis
- ± pathological fx (up to 10% reported)
- Uncommonly (~10%) with intralesional densities
- Cortical thinning and trabeculation
- Bone scan: peripheral uptake
- Angiography: hypovascular lesions with localized regions of hypervascularity
- Fluid-fluid levels on MRI (85%) when blood within the cysts layer out below serum in the cysts and septations
- Has been reported in monozygotic twins
- Unusually may cross physeal plate
- Epiphyseal location has been reported
- Unusual location
- Bloody!, as a sponge or a honeycomb filled with (unclotted) blood and fibrous cyst-like walls
HISTOLOGIC AND MOLECULAR FEATURES
- Fibrous cyst-like walls (not always seen because curettage fragments the flimsy lining)
- Blood-filled spaces (may be washed out of most cavities by formalin)
- Fibrous stroma with moderate to exuberant cellularity, esp in the early phase
- May have intramural calcified chondroid (up to 30%) or slivers of osteoid or woven bone
- Osteoclast-like giant cells scattered in the walls in variable numbers
- 'Pseudosarcomatous' nature of rare lesions
- ABCs harbor translocation of TRE17/USP6** Transcriptionally upregulated
- TRE17 encodes ubiquitin-specific protease (USP)
- TRE17 encodes a TBC domain that mediates binding to Arf6 GTPase
- TRE17 induces expression of MMP-9 and MMP-10
- MMP-9 transcription induced through activation of nuclear factor-kappaB
- Mediated (in part) by GTPase RhoA and its effector kinase, ROCK
- Requires USP but not Arf6 binding
- Induces formation of tumors with ABC features
- SS18 rearrangement has also been described in addition to USP6
DIFFERENTIAL CLINICOPATHOLOGIC DIAGNOSIS
- Juxtacortical chondroma in periosteal lesions without periosteal rim, but with a sclerotic cortical margin
- Osteosarcoma, particularly telangiectatic and fibroblastic variants
- Fibrous dysplasia
- Chondroblastoma with ABC changes
- CMF with ABC changes
DISEASE COURSE AND TREATMENT
- Never metastasizes, although LR (0-59% reported) may occur (esp skeletally immature pts)
- Median time to LR = 17 mos, rare > 2 yrs
- LR after curettage/burring/cementation ?5-17%
- LR after curettage/bone graft ?20-50% (only one had burring)
- LR after curettage and adjunctive cryotheraphy ~4-18%, adjunctive phenolization ~12-20%
- Cryotherapy LR of 5% reported successfully txd with 2nd curettage and cryotherapy
- (Wide cortical saucerization with curettage, cryotherapy or phenolization recommended tx)
- LR after marginal excision reported 0%
- Spontaneous healing has been reported (rare)
- Usually growth arrest occurs when epiphysis is involved (and has been reported without direct physeal involve-ment)
- Juxtaphyseal lesions may be treated with curettage and expectation of normal physeal growth reported
- Subperiosteal resection with reconstitution of fibula has been reported
- Spinal lesions (?20% of locations) may compress nerve roots or spinal cord
- En bloc excision for small tubular bones of the hand described with 8% LR
- Cavitational ultrasonic surgical aspirator (Cavitron) has been used to fascilitate resection
- 10% LR within 10 yrs, 2nd LR in 16.7% within 9 yrs
- Preoperative selective arterial embolization, complete curettage, bone grafting, ? fusion
- XRT has been used in spinal lesions, but is contraindicated now because of postradiation sarcoma
- Embolization alone after diagnostic bx can be curative, hastening maturation of the lesion
- Percutaneous repetitive sclerotherapy** Polidocanol is used
- Used successfully in pelvic and spine lesions
- Percutaneous CT-guided injection of DBM mixed with bone marrow concentrate reported in sacral lesions
- Percutaneous embolization(s) with alcohlic solution of Zein reported successful unless marked venous drainage apparent
- Postoperative fx in up to 10% reported
SOLID ABC VARIANT
- Rarely reported in the spine
- Lesion may have little or no areas of blood filled spaces, with areas of exuberant spindle cells
- Osteoid or chondroid may mislead to a dx of OSA
- Reparative nature of the lesion indicated by mature areas of hypocellularity
- Osteoclast-like giant cells and lymphocytes may be seen
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