Aneurysmal bone cyst

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Editor-In-Chief: C. Michael Gibson, M.S., M.D. [1]; Associate Editor(s)-in-Chief: Rohan A. Bhimani, M.B.B.S., D.N.B., M.Ch.[2]

Overview

Aneurysmal bone cyst (ABC) accounts for 1% to 2% of all primary bone tumors. The incidence of aneurysmal bone cyst is approximately 0.14 per 100,000 individuals worldwide. Adolescents and children are most affected by aneurysmal bone cyst. The age distribution of aneurysmal bone cyst is between 1.5-25 years. The mean age of the patients with aneurysmal bone cyst is 16 years. Women are more commonly affected than men, with a 1:0.84 ratio. In 1942, Jaffe and Lichtenstein first described aneurysmal bone cyst. Aneurysmal bone cyst can be classified based on mechanism of origin into primary and secondary. The true causes of aneurysmal bone cyst are not known. ABCs usually present in the long bones such as the humerus, femur, tibia and fibula. About 25% of ABC's are seen in posterior element of spine. ABC's typically occur in the metaphysis of the long bones. The hallmark of ABC is Pain, swelling and pathological fracture. The mainstay of treatment for ABC is surgery in form of curettage and bone grafting.

Historical Perspective

  • In 1942, Jaffe and Lichtenstein first described aneurysmal bone cyst.[1][2]
  • In 1940, Ewing propoosed that aneurysmal bone cyst was caused by increased venous pressure.
  • In 1956, Cruz and Coley also supported Ewing's hypothesis.
  • In 1956, Barnes hypothesized that aneurysmal bone cyst was due to an abnormal response to injury.
  • In 1956, Otterdoom after a thorough pathological study, concluded that aneurysmal bone cyst was an angioma of bone.

Classification

  • Aneurysmal bone cyst(ABC) can be classified based on imaging findings and on mechanism of origin.

Mechanism of Origin

Based on mechanism of origin, aneurysmal bone cyst can be classified into:[3][4]

Primary Aneurysmal Bone Cyst(ABC)

  • When ABC arises de novo, it is called primary ABC.
  • About 70% of ABC's are primary in origin.

Secondary Aneurysmal Bone Cyst(ABC)

Enneking (MSTS) Staging System

Stages Description
1 Latent: Well demarcated borders
2 Active: Indistinct borders
3 Aggressive: Indistinct borders

Pathophysiology

  • The exact pathogenesis of aneurysmal bone cyst is not fully understood.[6][7]
  • Various theories have been proposed concerning the pathogenesis of aneurysmal bone cyst:
    • Local alteration in hemodynamics resulting in markedly increased venous pressure and development of a dilated vascular bed within the involved bone.[8][9]
    • Primary ABCs demonstrate a t(16;17)(q22;p13) fusion of the TRE17/CDH11-USP6 oncogene leading to increased cellular cadherin-11 activity which arrests osteoblastic maturation in a more primitive state.[10][11][12]
  • ABCs usually present in the long bones such as the humerus, femur, tibia; and fibula.
  • About 25% of ABC's are seen in posterior element of spine.
  • ABC's typically occur in the metaphysis of the long bones.

Causes

Differentiating Aneurysmal Bone Cyst from Other Diseases

Aneurysmal bone cyst must be differentiated from following bone disorders:

Disease Bubbly lytic lesion on x-ray Lakes of Blood on histology Diagnosis Treatment is curretage and bone grafting
Aneurysmal bone cyst + + Radiology and biopsy +
Unicameral bone cyst + - Radiology and biopsy -
Non ossifying fibroma + - Radiology and biopsy -
Giant cell tumor - - Radiology and Biopsy +
Chondroblastoma - - Biopsy +
Chondromyxoid Fibroma - - Radiology and biopsy +
Osteoblastoma - - Radiology and biopsy +
Telangiectatic osteosarcoma - + Radiology and biopsy -

Epidemiology and Demographics

Risk Factors

There are no established risk factors for aneurysmal bone cyst.

Screening

There is insufficient evidence to recommend routine screening for aneurysmal bone cyst.

Natural History, Complications, and Prognosis

Diagnosis

Diagnostic Study of Choice

History and Symptoms

Physical Examination

X-ray showing aneurysmal bone cyst of left proximal femur.Source: Case courtesy of Dr Lawrence Chia Wei Oh, Radiopaedia.org, rID: 28998
  • Common physical examination findings of aneurysmal bone cyst include:
    • Deformity
    • Decreased range of motion, weakness, or stiffness
    • Torticollis
    • Occasionally, bruit over the affected area
    • Warmth over the affected area

Laboratory Findings

There are no diagnostic laboratory findings associated with aneurysmal bone cyst.

Electrocardiogram

There are no ECG findings associated with aneurysmal bone cyst.

X-ray

CT scan showing aneurysmal bone cyst of posterior element of spine.Source: Case courtesy of RMH Neuropathology, Radiopaedia.org, rID: 29294
  • Radiologically aneurysmal bone cyst can be stratified into 4 phases:[23]
    • Initial phase 1 – Small, lytic lesion without evidence of lifting off of the periosteum.
    • Phase 2 - Rapidly enlarging demonstrating a characteristic blowout appearance.
    • Phase 3 - Growth slows or stops after some sort of intervention.
    • Phase 4 - Healing with progressive ossification and calcification of the cavity.

Echocardiography or Ultrasound

There are no echocardiography/ultrasound findings associated with aneurysmal bone cyst.

CT scan

  • CT scan confirms the x-ray findings.[24]
  • In addition, CT demonstrates internal septation such as calcified rim, giving an eggshell appearance, which may be completely or partially intact.
  • Fluid-fluid levels can also be seen.

MRI

MRI showing aneurysmal bone cyst of left proximal femur.Source: Case courtesy of Dr Lawrence Chia Wei Oh, Radiopaedia.org, rID: 28998

Other Imaging Findings

Bone Scan

  • Increased uptake is observed around the lesion of aneurysmal bone cyst.
  • Findings often demonstrate a halo effect of increased radionuclide uptake surrounding an area of little uptake.

Other Diagnostic Studies

Angiography

  • Angiography demonstrates a hypervascular area around the aneurysmal bone cyst.
  • An intense diffuse area of persistent contrast accumulation may be visualized without main afferent or efferent vessels observed.
  • It can be helpful to plan selective arterial embolization as the primary treatment or as a preoperative method to help control intraoperative blood loss.

Treatment

Medical Therapy

There is no treatment for aneurysmal bone cyst, the mainstay of therapy is surgery.

Surgery

Aggressive curettage with adjuvant and bone grafting

Indications

  • Symptomatic aneurysmal bone cyst without acute fracture

Technique

Recurrence

  • Local recurrence in up to 25% and more common in children with open ephyses.

Primary Prevention

There are no established measures for the primary prevention of aneurysmal bone cyst.

Secondary Prevention

There are no established measures for the secondary prevention of aneurysmal bone cyst.

References

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  2. GODFREY LW, GRESHAM GA (1959). "The natural history of aneurysmal bone cyst". Proc R Soc Med. 52: 900–5. PMC 1870818. PMID 13850355.
  3. Feigenberg SJ, Marcus RB, Zlotecki RA, Scarborough MT, Berrey BH, Enneking WF (2001). "Megavoltage radiotherapy for aneurysmal bone cysts". Int J Radiat Oncol Biol Phys. 49 (5): 1243–7. PMID 11286830.
  4. Lin PP, Brown C, Raymond AK, Deavers MT, Yasko AW (2008). "Aneurysmal bone cysts recur at juxtaphyseal locations in skeletally immature patients". Clin Orthop Relat Res. 466 (3): 722–8. doi:10.1007/s11999-007-0080-8. PMC 2505199. PMID 18202898.
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  6. Peabody, Terrance (2014). Orthopaedic oncology : primary and metastatic tumors of the skeletal system. Cham: Springer. ISBN 9783319073224.
  7. Cottalorda J, Bourelle S (2007). "Modern concepts of primary aneurysmal bone cyst". Arch Orthop Trauma Surg. 127 (2): 105–14. doi:10.1007/s00402-006-0223-5. PMID 16937137.
  8. LICHTENSTEIN L (1953). "Aneurysmal bone cyst; further observations". Cancer. 6 (6): 1228–37. PMID 13106840.
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  10. Panoutsakopoulos G, Pandis N, Kyriazoglou I, Gustafson P, Mertens F, Mandahl N (1999). "Recurrent t(16;17)(q22;p13) in aneurysmal bone cysts". Genes Chromosomes Cancer. 26 (3): 265–6. PMID 10502326.
  11. Oliveira AM, Perez-Atayde AR, Dal Cin P, Gebhardt MC, Chen CJ, Neff JR; et al. (2005). "Aneurysmal bone cyst variant translocations upregulate USP6 transcription by promoter swapping with the ZNF9, COL1A1, TRAP150, and OMD genes". Oncogene. 24 (21): 3419–26. doi:10.1038/sj.onc.1208506. PMID 15735689.
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  20. Yamamoto T, Nagira K, Akisue T, Marui T, Hitora T, Kawamoto T; et al. (2003). "Fine-needle aspiration biopsy of solid aneurysmal bone cyst in the humerus". Diagn Cytopathol. 28 (3): 159–62. doi:10.1002/dc.10248. PMID 12619099.
  21. Martinez V, Sissons HA (1988). "Aneurysmal bone cyst. A review of 123 cases including primary lesions and those secondary to other bone pathology". Cancer. 61 (11): 2291–304. PMID 2835141.
  22. Nobler MP, Higinbotham NL, Phillips RF (1968). "The cure of aneurysmal bone cyst. Irradiation superior to surgery in an analysis of 33 cases". Radiology. 90 (6): 1185–92. doi:10.1148/90.6.1185. PMID 5656741.
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  25. Boubbou M, Atarraf K, Chater L, Afifi A, Tizniti S (2013). "Aneurysmal bone cyst primary--about eight pediatric cases: radiological aspects and review of the literature". Pan Afr Med J. 15: 111. doi:10.11604/pamj.2013.15.111.2117. PMC 3828064. PMID 24244797.
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