Skip to main content

Advertisement

Log in

Aneurysmal bone cyst of the spine treated by concentrated bone marrow: clinical cases and review of the literature

  • Case Report
  • Published:
European Spine Journal Aims and scope Submit manuscript

Abstract

Purpose

ABC is a benign lesion with unpredictable behavior. Its treatment is challenging, especially in poorly accessible surgical areas, such as spine and pelvis. Currently, the first-line treatment of ABC is repeated selective arterial embolization (SAE) until healing. Other options have been used with variable success rates. We propose an alternative treatment for spine aneurysmal bone cyst (sABC) based on the injection of concentrated autologous bone marrow.

Methods

We retrospectively report and analyze here two cases of patients, a 14-year-old girl and a 16-year-old boy, both affected by ABC in C2 vertebra which were impossible to treat by SAE. They were treated with single or repeated injection of concentrated autologous bone marrow into the lesion. Their follow-up period is 27 months for both patients.

Results

In the two cases reported here we observed a progressive ossification of the lesion, which was slow in one case, requiring three subsequent injections of concentrated bone marrow, and fast in the other case, beginning 1 month after the procedure. In both cases, the healing of the lesion was associated with symptom relief and the clinical status of the patients remains stable after 2 years.

Conclusions

Although SAE can still be considered the first line in the treatment of ABC in the axial skeleton, new promising therapeutic procedures involving the use of mesenchymal stem cells are developing.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Fig. 1
Fig. 2
Fig. 3
Fig. 4
Fig. 5
Fig. 6

References

  1. Mascard E, Gomez-Brouchet A, Lambot K (2015) Bone cysts: unicameral and aneurismal bone cyst. Orthop Traumatol Surg Res 101(1 Suppl):S119–S127

    Article  CAS  PubMed  Google Scholar 

  2. Enneking WF, Spanier SS, Goodman MA (1980) A system for the surgical staging of musculoskeletal sarcoma. Clin Orthop Relat Res 153:106–120

    Google Scholar 

  3. Enneking WF (1986) A system of staging musculoskeletal neoplasms. Clin Orthop Relat Res 204:9–24

    Google Scholar 

  4. Boriani S, De Iure F, Campanacci L, Gasbarrini A, Bandiera S, Biagini R, Bertoni F, Picci P (2001) Aneurysmal bone cyst of the mobile spine: report on 41 cases. Spine 26:27–35

    Article  CAS  PubMed  Google Scholar 

  5. Boriani S, Bandiera S, Donthineni R, Amendola L, Cappuccio M, De Iure F, Gasbarrini A (2010) Morbidity of en bloc resections in the spine. Eur Spine J 19:231–241

    Article  PubMed  Google Scholar 

  6. Boriani S, Lo SF, Puvanesarajah V, Fisher CG, Varga PP, Rhines LD, Germscheid NM, Luzzati A, Chou D, Reynolds JJ, Williams RP, Zadnik P, Groves M, Sciubba DM, Bettegowda C, Gokaslan ZL (2014) AOSpine knowledge forum tumor. Aneurysmal bone cysts of the spine: treatment options and considerations. J Neurooncol 120:171–178

    Article  PubMed  Google Scholar 

  7. Amendola L, Simonetti L, Simoes CE, Bandiera S, De Iure F, Boriani S (2013) Aneurysmal bone cyst of the mobile spine: the therapeutic role of embolization. Eur Spine J 22:533–541

    Article  CAS  PubMed  Google Scholar 

  8. Di Bella C, Dozza B, Frisoni T, Cevolani L, Donati D (2010) Injection of demineralized bone matrix with bone marrow concentrate improves healing in unicameral bone cyst. Clin Orthop Relat Res 468:3047–3055

    Article  PubMed  PubMed Central  Google Scholar 

  9. Bulgin D, Irha E, Hodzic E, Nemec B (2013) Autologous bone marrow derived mononuclear cells combined with β-tricalcium phosphate and absorbable atelocollagen for a treatment of aneurysmal bone cyst of the humerus in child. J Biomater Appl 28:343–353

    Article  PubMed  Google Scholar 

  10. Boriani S, Weinstein JN, Biagini R (1997) Primary bone tumors of the spine. Terminology and surgical staging. Spine (Phila Pa 1976) 22:1036–1044 (review)

  11. Bilsky MH, Laufer I, Fourney DR, Groff M, Schmidt MH, Varga PP, Vrionis FD, Yamada Y, Gerszten PC, Kuklo TR (2010) Reliability analysis of the epidural spinal cord compression scale. J Neurosurg Spine 13:324–328

    Article  PubMed  Google Scholar 

  12. Jaffe H, Lichtenstein L (1942) Solitary unicameral bone cyst: with emphasis on the roentgen picture, the pathologic appearance and the pathogenesis. Arch Surg 44:1004–1025

    Article  Google Scholar 

  13. Lichtenstein L (1950) Aneurysmal bone cyst. A pathological entity commonly mistaken for giant-cell tumor and occasionally for hemangioma and osteogenic sarcoma. Cancer 3:279–289

    Article  Google Scholar 

  14. Leithner A, Windhager R, Lang S, Haas OA, Kainberger F, Kotz R (1999) Aneurysmal bone cyst. A population based epidemiologic study and literature review. Clin Orthop Relat Res 363:176–179

    Article  Google Scholar 

  15. Karadeniz E (2013) Aneurysmal bone cyst-like areas as a sign of metastatic disease in the spinal column. Acta Orthop Traumatol Turc 47:366–369

    Article  PubMed  Google Scholar 

  16. Bandiera S, Bacchini P, Bertoni F (2000) Secondary aneurysmal bone cyst simulating malignant transformation in fibrous dysplasia. Orthopedics 23:1205–1207

    CAS  PubMed  Google Scholar 

  17. Boriani S, Biagini R, De Iure F, Andreoli I, Campanacci L, De Fiore M, Zanoni A (1995) Primary bone tumors of the spine: a survey of the evaluation and treatment at the Istituto Ortopedico Rizzoli. Orthopedics 18:993–1000

    CAS  PubMed  Google Scholar 

  18. Tillman BP, Dahlin DC, Lipscomb PR, Stewart JR (1968) Aneurysmal bone cyst: an analysis of ninety-five cases. Mayo Clin Proc 43:478–495

    CAS  PubMed  Google Scholar 

  19. Hay MC, Paterson D, Taylor TK (1978) Aneurysmal bone cysts of the spine. J Bone Jt Surg Br 60-B:406–411

  20. Ameli NO, Abbassioun K, Saleh H, Eslamdoost A (1985) Aneurysmal bone cysts of the spine. Report of 17 cases. J Neurosurg 63:685–690

    Article  CAS  PubMed  Google Scholar 

  21. Capanna R, Albisinni U, Picci P, Calderoni P, Campanacci M, Springfield DS (1985) Aneurysmal bone cyst of the spine. J Bone Jt Surg Am 67:527–531

    Article  CAS  Google Scholar 

  22. De Kleuver M, van der Heul RO, Veraart BE (1998) Aneurysmal bone cyst of the spine: 31 cases and the importance of the surgical approach. J Pediatr Orthop B 7:286–292

    Article  PubMed  Google Scholar 

  23. Papagelopoulos PJ, Currier BL, Shaughnessy WJ, Sim FH, Ebsersold MJ, Bond JR, Unni KK (1998) Aneurysmal bone cyst of the spine. Management and outcome. Spine (Phila Pa 1976) 23:621–628

  24. Campanacci M, Boriani S, Savini R (1990) Staging, biopsy, surgical planning of primary spinal tumors. Chir Organ Mov 75:99–103

    CAS  Google Scholar 

  25. Biesecker JL, Marcove RC, Huvos AG, Miké V (1970) Aneurysmal bone cysts. A clinicopathologic study of 66 cases. Cancer 26:615–625

    Article  CAS  PubMed  Google Scholar 

  26. Koskinen EV, Visuri TI, Holmström T, Roukkula MA (1976) Aneurysmal bone cyst: evaluation of resection and of curettage in 20 cases. Clin Orthop Relat Res 118:136–146

    Google Scholar 

  27. Campanacci M, Cervellati C, Donati U, Bertoni F (1976) Aneurysmal bone cyst (a study of 127 cases, 72 with longterm follow up). Ital J Orthop Traumatol 2:341–353

    CAS  PubMed  Google Scholar 

  28. Ruiter DJ, van Rijssel TG, van der Velde EA (1977) Aneurysmal bone cysts: a clinicopathological study of 105 cases. Cancer 39:2231–2239

    Article  CAS  PubMed  Google Scholar 

  29. Martinez V, Sissons HA (1988) Aneurysmal bone cyst. A review of 123 cases including primary lesions and those secondary to other bone pathology. Cancer 61:2291–2304

    Article  CAS  PubMed  Google Scholar 

  30. Szendröi M, Cser I, Kónya A, Rényi-Vámos A (1992) Aneurysmal bone cyst. A review of 52 primary and 16 secondary cases. Arch Orthop Trauma Surg 111:318–322

    Article  PubMed  Google Scholar 

  31. Vergel De Dios AM, Bond JR, Shives TC, McLeod RA, Unni KK (1992) Aneurysmal bone cyst. A clinicopathologic study of 238 cases. Cancer 69:2921–2931

    Article  CAS  PubMed  Google Scholar 

  32. Mankin HJ, Hornicek FJ, Ortiz-Cruz E, Villafuerte J, Gebhardt MC (2005) Aneurysmal bone cyst: a review of 150 patients. J Clin Oncol 23:6756–6762

    Article  PubMed  Google Scholar 

  33. Bandiera S, Boriani S, Donthineni R, Amendola L, Cappuccio M, Gasbarrini A (2009) Complications of en bloc resections in the spine. Orthop Clin N Am 40:125–131 (vii)

  34. Zileli M, Isik HS, Ogut FE, Is M, Cagli S, Calli C (2013) Aneurysmal bone cysts of the spine. Eur Spine J 22:593–601

    Article  PubMed  Google Scholar 

  35. Boriani S (2013) Reviewer’s comment concerning “aneurysmal bone cysts of the spine” (doi:10.1007/s00586-012-2510-x by M. Zileli et al.). Eur Spine J 22:602–604

  36. Szendröi M, Antal I, Liszka G, Kónya A (1992) Calcitonin therapy of aneurysmal bone cysts. J Cancer Res Clin Oncol 19:61–65

    Article  Google Scholar 

  37. Scaglietti O, Marchetti PG, Bartolozzi P (1982) Final results obtained in the treatment of bone cysts with methylprednisolone acetate (depo-medrol) and a discussion of results achieved in other bone lesions. Clin Orthop Relat Res 165:33–42

    Google Scholar 

  38. Funayama T, Gasbarrini A, Ghermandi R, Girolami M, Boriani S (2016) Solitary bone cyst of a lumbar vertebra treated with percutaneous steroid injection: a case report and review of literature. Eur Spine J

  39. Shiels WE 2nd, Mayerson JL (2013) Percutaneous doxycycline treatment of aneurysmal bone cysts with low recurrence rate: a preliminary report. Clin Orthop Relat Res 471:2675–2683

    Article  PubMed  PubMed Central  Google Scholar 

  40. Cottalorda J, Bourelle S (2007) Modern concepts of primary aneurysmal bone cyst. Arch Orthop Trauma Surg 127:105–114

    Article  PubMed  Google Scholar 

  41. Turowski B, Schellhammer F, Herdmann J, Rommel F (2005) Fatal ethibloc embolization of vertebrobasilar system following percutaneous injection into aneurysmal bone cyst of the second cervical vertebra. Am J Neuroradiol 26:1883–1884

    PubMed  Google Scholar 

  42. Pauli C, Fuchs B, Pfirrmann C et al (2014) Response of an aggressive periosteal aneurysmal bone cyst (ABC) of the radius to denosumab therapy. World J Surg Oncol 12:17

    Article  PubMed  PubMed Central  Google Scholar 

  43. Lange T, Stehling C, Frohlich B et al (2013) Denosumab: a potential new and innovative treatment option for aneurysmal bone cysts. Eur Spine J 22:1417–1422

    Article  PubMed  PubMed Central  Google Scholar 

  44. Pelle DW, Ringler JW, Peacock JD et al (2014) Targeting receptor–activator of nuclear kappaB ligand in aneurysmal bone cysts: verification of target and therapeutic response. Transl Res 164:139–148

    Article  CAS  PubMed  Google Scholar 

  45. Skubitz KM, Peltola JC, Santos ER, Cheng EY (2015) Response of aneurysmal bone cyst to Denosumab. Spine (Phila Pa 1976) 40(22):E1201–E1204

  46. Dubory A, Missenard G, Domont J, Court C (2016) Interest of Denosumab for the treatment of giant-cells tumors and aneurysmal bone cysts of the spine. About nine cases. Spine (Phila Pa 1976) 41(11):E654–E660

  47. Ghermandi R, Terzi S, Gasbarrini A, Boriani S (2016) Denosumab: non-surgical treatment option for selective arterial embolization resistant aneurysmal bone cyst of the spine and sacrum. Case report. Eur Rev Med Pharmacol Sci 20(17):3692–3695

    CAS  PubMed  Google Scholar 

  48. Barbanti Brodano G, Terzi S, Trombi L, Griffoni C, Valtieri M, Boriani S, Magli MC (2013) Mesenchymal stem cells derived from vertebrae (vMSCs) show best biological properties. Eur Spine J 22(Suppl 6):S979–S984

    Article  PubMed  Google Scholar 

  49. Docquier P-L, Delloye C (2005) Treatment of aneurysmal bone cysts by introduction of demineralized bone and autogenous bone marrow. J Bone Jt Surg Am 87:2253–2258

    Google Scholar 

  50. Donati D, Frisoni T, Dozza B, DeGroot H, Albisinni U, Giannini S (2011) Advance in the treatment of aneurysmal bone cyst of the sacrum. Skelet Radiol 40:1461–1466

    Article  Google Scholar 

  51. Bivins E, Alidina JA, Bancroft LW (2015) Aneurysmal bone cyst involving the C2 vertebra. Orthopedics 38(78):141–143

    Google Scholar 

Download references

Acknowledgements

The authors thank Mr. Carlo Piovani for his helpful collaboration in patients’ images storage and editing and Dr. Cristiana Griffoni for collaboration in the manuscript editing.

Author information

Authors and Affiliations

Authors

Corresponding author

Correspondence to Giovanni Barbanti-Brodano.

Ethics declarations

Conflict of interest

The authors declare that they have no conflict of interest.

Ethical approval

All procedures were in accordance with the ethical standards of the Institutional Research Committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards. Considering this is a retrospective study, formal consent is not required.

Rights and permissions

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Barbanti-Brodano, G., Girolami, M., Ghermandi, R. et al. Aneurysmal bone cyst of the spine treated by concentrated bone marrow: clinical cases and review of the literature. Eur Spine J 26 (Suppl 1), 158–166 (2017). https://doi.org/10.1007/s00586-017-4978-x

Download citation

  • Received:

  • Revised:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00586-017-4978-x

Keywords

Navigation