Abstract
Multifocal, extraosseous, and surface aneurysmal bone cysts are rare variants of the primary lesions. The clinicopathological features are similar, and the optimal treatment is surgical. Although local recurrences may occur, the prognosis is excellent. This review article introduces the readers to a rare diagnosis which they may have been previously unfamiliar with, presents the clinicopathological and imaging features of these rare aneurysmal bone cyst variants, and discusses their diagnosis and treatment. The clinicians who treat patients with aneurysmal bone cysts should be familiar with these uncommon entities and their differential diagnosis.
Similar content being viewed by others
References
Capanna R, Campanacci DA, Manfrini M (1996) Unicameral and aneurysmal bone cysts. Orthop Clin N Am 27(3):605–614
Cottalorda J, Bourelle S (2007) Modern concepts of primary aneurysmal bone cyst. Arch Orthop Trauma Surg 127(2):105–114
Mascard E, Gomez-Brouchet A, Lambot K (2015) Bone cysts: unicameral and aneurysmal bone cyst. Orthop Traumatol Surg Res OTSR 101(1 Suppl):S119–S127
Mavrogenis AF, Angelini A, Rossi G, Rimondi E, Guerra G, Ruggieri P (2014) Successful NBCA embolization of a T2 aneurysmal bone cyst. Acta Orthop Belg 80(1):126–131
Mavrogenis AF, Rossi G, Rimondi E, Ruggieri P (2011) Aneurysmal bone cyst of the acromion treated by selective arterial embolization. J Pediatr Orthop B 20(5):354–358
Mavrogenis AF, Rossi G, Rimondi E, Ruggieri P (2012) Successful treatment of aneurysmal bone cyst of the proximal fibula with embolization. Eur J Orthop Surg Traumatol Orthop Traumatol 22(Suppl 1):199–204
Mavrogenis AF, Skarpidi E, Papagelopoulos PJ (2010) Solid variant of aneurysmal bone cyst of the hamate. Musculoskelet Surg 94(3):145–150
Rapp TB, Ward JP, Alaia MJ (2012) Aneurysmal bone cyst. J Am Acad Orthop Surg 20(4):233–241
Rossi G, Angelini A, Mavrogenis AF, Rimondi E, Ruggieri P (2010) Successful treatment of aneurysmal bone cyst of the hip in a child by selective transcatheter arterial embolization. J Vasc Interv Radiol JVIR 21(10):1591–1595
Rossi G, Mavrogenis AF, Facchini G, Bartalena T, Rimondi E, Renzulli M et al (2017) How effective is embolization with N-2-butyl-cyanoacrylate for aneurysmal bone cysts? Int Orthop 41(8):1685–1692
Rossi G, Mavrogenis AF, Papagelopoulos PJ, Rimondi E, Ruggieri P (2012) Successful treatment of aggressive aneurysmal bone cyst of the pelvis with serial embolization. Orthopedics 35(6):e963–e968
Rossi G, Mavrogenis AF, Rimondi E, Ciccarese F, Tranfaglia C, Angelelli B et al (2011) Selective arterial embolisation for bone tumours: experience of 454 cases. Radiol Med (Torino) 116(5):793–808
Tournis S, Balanika A, Megaloikonomos PD, Mavrogenis AF (2017) Secondary aneurysmal bone cyst in McCune-Albright syndrome. Clin Cases Miner Bone Metab Off J Ital Soc Osteoporos Miner Metab Skelet Dis 14(3):332–335
Dal Cin P, Kozakewich HP, Goumnerova L, Mankin HJ, Rosenberg AE, Fletcher JA (2000) Variant translocations involving 16q22 and 17p13 in solid variant and extraosseous forms of aneurysmal bone cyst. Genes Chromosom Cancer 28(2):233–234
Althof PA, Ohmori K, Zhou M, Bailey JM, Bridge RS, Nelson M et al (2004) Cytogenetic and molecular cytogenetic findings in 43 aneurysmal bone cysts: aberrations of 17p mapped to 17p13.2 by fluorescence in situ hybridization. Modern Pathol Off J U S Can Acad Pathol Inc 17(5):518–525
Jaffe HL, Lichtenstein L (1942) Solitary unicameral bone cyst with emphasis on the roentgen picture, the pathologic appearances, and the pathogenesis. Arch Surg 44(6):1004–1025
Blackburn PR, Davila JI, Jackson RA, Fadra N, Atiq MA, Pitel BA et al (2019) RNA sequencing identifies a novel USP9X-USP6 promoter swap gene fusion in a primary aneurysmal bone cyst. Genes Chromosom Cancer 58(8):589–594
Kenney B, Richkind KE, Zambrano E (2007) Solid variant of aneurysmal bone cyst with a novel (X;9) translocation. Cancer Genet Cytogenet 178(2):155–159
Panoutsakopoulos G, Pandis N, Kyriazoglou I, Gustafson P, Mertens F, Mandahl N (1999) Recurrent t(16;17)(q22;p13) in aneurysmal bone cysts. Genes Chromosom Cancer 26(3):265–266
Mavrogenis AF, Angelini A, Errani C, Rimondi E (2014) How should musculoskeletal biopsies be performed? Orthopedics 37(9):585–588
Pietschmann MF, Oliveira AM, Chou MM, Ihrler S, Niederhagen M, Baur-Melnyk A et al (2011) Aneurysmal bone cysts of soft tissue represent true neoplasms: a report of two cases. JBJS Am 93(9):e45
Clough JR, Price CH (1973) Aneurysmal bone cyst: pathogenesis and long term results of treatment. Clin Orthop Relat Res 97:52–63
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):2291–2304
Kransdorf MJ, Sweet DE (1995) Aneurysmal bone cyst: concept, controversy, clinical presentation, and imaging. AJR Am J Roentgenol 164(3):573–580
Baruffi MR, Neto JB, Barbieri CH, Casartelli C (2001) Aneurysmal bone cyst with chromosomal changes involving 7q and 16p. Cancer Genet Cytogenet 129(2):177–180
Oliveira AM, Hsi BL, Weremowicz S et al (2004) USP6 (Tre2) fusion oncogenes in aneurysmal bone cyst. Cancer Res 64:1920–1923
Sciot R, Dorfman H, Brys P et al (2000) Cytogenetic-morphologic correlations in aneurysmal bone cyst, giant cell tumor of bone and combined lesions. A report from the CHAMP study group. Mod Pathol 13:1206–1210
Ye Y, Pringle LM, Lau AW et al (2010) TRE17/USP6 oncogene translocated in aneurysmal bone cyst induces matrix metalloproteinase production via activation of NF-kappaB. Oncogene 29:3619–3629
Masuda-Robens JM, Kutney SN, Qi H, Chou MM (2003) The TRE17 oncogene encodes a component of a novel effector pathway for Rho GTPases Cdc42 and Rac1 and stimulates actin remodeling. Mol Cell Biol 23(6):2151–2161
Capanna R, Betelli G, Biagini R, Ruggieri P, Bertoni F, Campanacci M (1985) Aneurysmal bone cysts of long bones. Ital J Orthop Traumatol 11(4):409–417
Okada K, Masuda H, Shozawa T, Arai M (1989) A small aneurysmal bone cyst restricted to the cortical bone of the femur resembling so-called subperiosteal giant cell tumor or subperiosteal osteoclasia. Acta Pathol Jpn 39(8):539–544
Geschickter CF, Copeland MM (1949) Subperiosteal giant cell tumor. Tumors of bone, 3rd edn. J.B. Lippincott, Philadelphia, pp 309–316
Ajilogba KA, Kaur H, Duncan R, McFarlane JH, Watt AJ (2005) Extraosseous aneurysmal bone cyst in a 12-year-old girl. Pediatr Radiol 35(12):1240–1242
Alraiyes TM, Stavropoulos NA, Alaseem A, Jung S, Turcotte RE (2018) Periosteal aneurysmal bone cyst. Lancet Oncol 19(4):e218
Amir G, Mogle P, Sucher E (1992) Case report 729. Myositis ossificans and aneurysmal bone cyst. Skelet Radiol 21(4):257–259
Baker KS, Gould ES, Patel HB, Hwang SJ (2015) Soft tissue aneurysmal bone cyst: a rare case in a middle aged patient. J Radiol Case Rep 9(1):26–35
Hao Y, Wang L, Yan M, Jin F, Ge S, Dai K (2012) Soft tissue aneurysmal bone cyst in a 10-year-old girl. Oncol Lett 3(3):545–548
Karkuzhali P, Bhattacharyya M, Sumitha P (2007) Multiple soft tissue aneurysmal cysts: an occurrence after resection of primary aneurysmal bone cyst of fibula. Indian J Orthop 41(3):246–249
Kobayashi S, Hayakawa K, Takeno K, Baba H, Meir A (2009) Parosteal aneurysmal bone cyst of the humerus with birdcage-like ossification on three-dimensional CT scanning: a case report. Joint Bone Spine Rev Rhum 76(6):705–707
Lopez LV, Rodriguez MG, Siegal GP, Wei S (2017) Extraskeletal aneurysmal bone cyst: report of a case and review of the literature. Pathol Res Pract 213(11):1445–1449
Lopez-Barea F, Rodriguez-Peralto JL, Burgos-Lizaldez E, Alvarez-Linera J, Sanchez-Herrera S (1996) Primary aneurysmal cyst of soft tissue. Report of a case with ultrastructural and MRI studies. Virchows Arch Int J Pathol 428(2):125–129
Muller CS, Kim YJ, Koch K, Schneider G, Pfohler C, Kohn D et al (2016) First report of an aneurysmal bone cyst presenting as subungual mass. J Cutan Pathol 43(8):711–716
Nielsen GP, Fletcher CD, Smith MA, Rybak L, Rosenberg AE (2002) Soft tissue aneurysmal bone cyst: a clinicopathologic study of five cases. Am J Surg Pathol 26(1):64–69
Petrik PK, Findlay JM, Sherlock RA (1993) Aneurysmal cyst, bone type, primary in an artery. Am J Surg Pathol 17(10):1062–1066
Rodriguez-Peralto JL, Lopez-Barea F, Sanchez-Herrera S, Atienza M (1994) Primary aneurysmal cyst of soft tissues (extraosseous aneurysmal cyst). Am J Surg Pathol 18(6):632–636
Sahu A, Gujral SS, Gaur S (2008) Extraosseous aneurysmal cyst in hand: a case report. Cases J 1(1):268
Salm R, Sissons HA (1972) Giant-cell tumours of soft tissues. J Pathol 107(1):27–39
Samura H, Shiraishi M, Tokashiki H, Nosato E, Miyazato H, Muto Y (2000) An extraosseous aneurysmal cyst in the pelvic cavity: report of a case. Clin Imaging 24(2):68–71
Shannon P, Bedard Y, Bell R, Kandel R (1997) Aneurysmal cyst of soft tissue: report of a case with serial magnetic resonance imaging and biopsy. Human Pathol 28(2):255–257
Sukov WR, Franco MF, Erickson-Johnson M, Chou MM, Unni KK, Wenger DE et al (2008) Frequency of USP6 rearrangements in myositis ossificans, brown tumor, and cherubism: molecular cytogenetic evidence that a subset of “myositis ossificans-like lesions” are the early phases in the formation of soft-tissue aneurysmal bone cyst. Skelet Radiol 37(4):321–327
Wang XL, Gielen JL, Salgado R, Delrue F, De Schepper AM (2004) Soft tissue aneurysmal bone cyst. Skelet Radiol 33(8):477–480
Woertler K, Brinkschmidt C (2002) Imaging features of subperiosteal aneurysmal bone cyst. Acta Radiol 43(3):336–339
Amer HZ, Mayerson JL, Klein MJ, Baker PB (2012) Metachronous aneurysmal bone cyst in a 9-year-old boy: case report and review of the literature. J Pediatr Orthop Part B 21(2):187–191
Donigan JA, Kebaish KM, McCarthy EF (2003) Metachronous aneurysmal bone cysts with involvement of the humerus and the thoracic vertebrae. Skelet Radiol 32(8):468–471
Niemeier TE, Leddy LR, Chapin RW, Smith TM (2013) Metachronous aneurysmal bone cysts in a fourteen-year-old girl: a case report and review of the literature. JBJS Case Connect 3(2 Suppl 8):1–7
Scheil-Bertram S, Hartwig E, Bruderlein S, Melzner I, von Baer A, Roessner A et al (2004) Metachronous and multiple aneurysmal bone cysts: a rare variant of primary aneurysmal bone cysts. Virchows Arch Int J Pathol 444(3):293–299
Sundaram M, McDonald DJ, Steigman CK, Bocchini T (1997) Metachronous multiple aneurysmal bone cysts. Skelet Radiol 26(9):564–567
Ellison DA, Sawyer JR, Parham DM, Nicholas R Jr (2007) Soft-tissue aneurysmal bone cyst: report of a case with t(5;17)(q33;p13). Pediatr Dev Pathol 10(1):46–49
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
Hudson TM (1984) Scintigraphy of aneurysmal bone cysts. AJR Am J Roentgenol 142(4):761–765
Saifuddin A, Burnett SJ, Mitchell R (1998) Pictorial review: ultrasonography of primary bone tumours. Clin Radiol 53(4):239–246
Meana Moris AR, Garcia Gonzalez P, Fuente Martin E, Gonzalez Suarez C, Moro Barrero L (2010) Primary giant cell tumor of soft tissue: fluid-fluid levels at MRI. Eur Radiol 20(6):1539–1543
Bu An S, Choi JA, Chung JH, Oh JH, Kang HS (2008) Giant cell tumor of soft tissue: a case with atypical US and MRI findings. Korean J Radiol 9(5):462–465
Maiya S, Davies M, Evans N, Grimer J (2002) Surface aneurysmal bone cysts: a pictorial review. Eur Radiol 12(1):99–108
Van Royen A, Vanhoenacker F, De Roeck J (2015) Surface aneurysmal bone cyst. J Belg Soc Radiol 99(2):13–15
Yalcinkaya M, Lapcin O, Arikan Y, Aycan OE, Ozer D, Kabukcuoglu YS (2016) Surface aneurysmal bone cyst: clinical and imaging features in 10 new cases. Orthopedics 39(5):e897–e903
Sherman RS, Soong KY (1957) Aneurysmal bone cyst: its roentgen diagnosis. Radiology 68(1):54–64
Funding
No benefits have been or will be received from a commercial party related directly or indirectly to the subject matter of this article.
Author information
Authors and Affiliations
Corresponding author
Ethics declarations
Conflict of interest
All authors declare that they have no conflict of interest.
Ethical approval
All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki Declaration and its later amendments or comparable ethical standards.
Informed consent
Informed consent was obtained from all individual participants included in the study.
Additional information
Publisher's Note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Rights and permissions
About this article
Cite this article
Angelini, A., Mavrogenis, A.F., Pagliarini, E. et al. Rare aneurysmal bone cysts: multifocal, extraosseous, and surface variants. Eur J Orthop Surg Traumatol 30, 969–978 (2020). https://doi.org/10.1007/s00590-020-02640-3
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s00590-020-02640-3