Skip to main content

Advertisement

SpringerLink
Log in

Paget’s sarcoma of the patella

  • Case Report
  • Published:
Skeletal Radiology Aims and scope Submit manuscript

Abstract

Paget’s sarcoma is a rare complication of Paget’s disease and isolated Paget’s disease of the patella is extremely rare. We describe a unique case of Paget’s sarcoma of the patella in a 69-year-old male farmer who had a remote history of a fracture in the same patella 40 years previously. In this case, imaging and pathogenesis of Paget’s disease of bone is described and factors implicated in the development of Paget’s disease in this patient are evaluated.

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

Access this article

Log in via an institution

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

Instant access to the full article PDF.

Institutional subscriptions

Fig. 1
Fig. 2
Fig. 3

References

  1. Ralston SH. Pathogenesis of Paget’s disease of bone. Bone. 2008;43:819–25.

    Article  CAS  PubMed  Google Scholar 

  2. Sun SG, Lau YS, Itonaga I, Sabokbar A, Athanasou NA. Bone stromal cells in pagetic bone and Paget’s sarcoma express RANKL and support human osteoclast formation. J Pathol. 2006;209:114–20.

    Article  CAS  PubMed  Google Scholar 

  3. Ralston SH. Paget’s disease of bone. N Engl J Med. 2013;368:644–50.

    Article  CAS  PubMed  Google Scholar 

  4. Solomon LR. Billiard players fingers: an unusual case of Paget disease of bone. BMJ. 1979;4:931.

    Article  Google Scholar 

  5. Gasper TM. Paget’s disease in a treadle machine operator. BMJ. 1979;5:1217–8.

    Article  Google Scholar 

  6. Mangham DC, Davie MW, Grimer RJ. Sarcoma arising in Paget’s disease of bone: declining incidence and increasing age at presentation. Bone. 2009;44:431–6.

    Article  CAS  PubMed  Google Scholar 

  7. Dickson DD, Camp JD, Ghormley RK. Osteitis deformans: Paget’s disease of the bone. Radiology. 1945;44:449.

    Article  Google Scholar 

  8. Gutman AB, Kasabach H. Paget’s disease (osteitis deformans). Am J Med Sci. 1936;191:361.

    Article  Google Scholar 

  9. Pyper JB. A case of Paget’s disease of the patella. Br Med J. 1958;1:267.

    Article  PubMed Central  CAS  PubMed  Google Scholar 

  10. Gordon L. Paget’s disease of the patella. J Bone Joint Surg. 1958;40A(6):1423–5.

    Google Scholar 

  11. Weinert CR, Wiss DA. Paget’s disease of the patella. Clin Orthop Relat Res. 1979;142:138–40.

    Google Scholar 

  12. D’Haem RJ, Cole RL. The case of the pathetic pagetic patella. Clin Nucl Med. 1989;14(4):306–8.

    Article  PubMed  Google Scholar 

  13. Stull MA, Moser RP, Vinh TN, Kransdorf MJ, Callaghan JJ. Paget’s disease of the patella. Skelet Radiol. 1990;19:407–10.

    Article  CAS  Google Scholar 

  14. Kleinbart FA, Vigorita VJ, Scalafani SJ, Bryk E. Am J Knee Surg. 1992;5(2):91–3.

    Google Scholar 

  15. Abamor E, Kitapci MT, Cila E, Gokcora N, Uluoglu O. Increased accumulation of Tl-201 in monostotic Paget’s disease of the patella. Clin Nucl Med. 2001;26(7):615–8.

    Article  CAS  PubMed  Google Scholar 

  16. Amr R, Choudry Q. Monostotic Paget’s disease of the patella. Bone. 2006;39(6):1382–6.

    Article  CAS  PubMed  Google Scholar 

  17. Ploumis A, Christodoulou P, Vrangalas V, Milias S, Pantazis E. Isolated Paget’s disease of the patella complicated with ipsilateral femoral head bone marrow oedema: a case report and review of the literature. South Med J. 2008;101(7):753–5.

    Article  PubMed  Google Scholar 

  18. Hernandez-Pascual C, Villanueva-Martinez M, Rios-Luna A, Benito-de Carmen F, Castelorio R. Monostotic patellar Paget’s disease. Acta Orth Mex. 201; 24(1): 14–17. [Article in Spanish]

  19. Streda A. Isolated Paget disease of the patella. Radiol Diagn. 1963;4:83–4. [Article in German].

    CAS  Google Scholar 

  20. Arnold H. Further case of Paget’s disease of patella. Z Arztl Fortbild. 1957;51(8):331–3 [Article in German].

    CAS  Google Scholar 

  21. von Hunziker H. Post traumatic osteosclerosis of the patella of the type of osteitis deformans of Paget. Schweiz Z Allg Pathol. 1947;10:654–66. [Article in German].

    Google Scholar 

  22. Busanelli T, Martuzzi M. Monostotic Paget’s disease localized on the patella. Chir Organi Mov. 1956;43(6):501–7. [Article in Italian].

    CAS  PubMed  Google Scholar 

  23. Lur’e AM, Sal’man MM. Roentgenologic diagnosis of Paget’s quiet necrosis of the patella. Vestn Rentgenol Radiol. 1955; (3):55–9. [Article in Russian]

  24. Wick MR, Siegal GP, Unni KK, McLeod RA, Greditzer HG. Sarcomas of bone complicating osteitis deformans (Paget’s disease). Am J Surg Pathol. 1981;51(1):47–59.

    Article  Google Scholar 

  25. Lopez C, Thomas DV, Davies AM. Neoplastic transformation and tumour-like lesions in Paget’s disease of bone: a pictorial review. Eur Radiol. 2003;13:151–63.

    Article  Google Scholar 

  26. Seton M. Paget disease of bone: diagnosis and drug therapy. Cleve Clin J Med. 2013;80(7):452–62.

    Article  PubMed  Google Scholar 

  27. Huvos AG, Butler A, Bretsky SS. Osteogenic sarcoma associated with Paget’s disease of bone: a clinicopathologic study of 65 patients. Cancer. 1983;52:1489–95.

    Article  CAS  PubMed  Google Scholar 

  28. Ruggieri P, Calabro T, Montalti M, Mercuri M. The role of surgery and adjuvants to survival in pagetic osteosarcoma. Clin Orthop Relat Res. 2010;468:2961–8.

    Google Scholar 

  29. Rendina D, Mossetti G, Soscia E, Sirignano C, Insabato L, Viceconti R, et al. Giant cell tumor and Paget’s disease of bone in one family: geographic clustering. Clin Orthop Relat Res. 2004;421:218–24.

    Article  PubMed  Google Scholar 

  30. Hadjipavlou A, Lander P, Srolovitz H, Enker IP. Malignant transformation in Paget disease of bone. Cancer. 1992;70(12):2802–8.

    Article  CAS  PubMed  Google Scholar 

  31. Whitten CR, Saifuddin A. MRI of Paget’s disease of bone. Clin Radiol. 2003;58:763–9.

    Article  CAS  PubMed  Google Scholar 

  32. Woo JH, Kim S, Choi SJ, Lee YH, Ji JD, Song GG. Diagnosis of Paget’s disease of the pelvis using F-18 FDG PET/CT. Int J Rheum Dis. 2010;13:e51–4.

    Article  PubMed  Google Scholar 

  33. Cook GJR, Maisey MN, Fogelman I. Fluorine-18-FDG PET in Paget’s disease of bone. J Nucl Med. 1997;38:1495–7.

    CAS  PubMed  Google Scholar 

  34. Sharma H, Jane MJ, Reid R. Scapulo-humeral Paget’s sarcoma: Scottish bone tumour registry experience. Eur J Cancer Care. 2005;14:367–72.

    Article  CAS  Google Scholar 

  35. Sugarbaker ED. Osteitis deformans (Paget’s disease of bones). Am J Surg. 1940;48:414.

    Article  Google Scholar 

  36. van Staa TP, Selby P, Leufken HGM, Lyles K, Sprafka JM, Cooper C. Incidence and natural history of Paget’s disease of bone in England and Wales. J Bone Miner Res. 2002;17(3):465–71.

    Article  PubMed  Google Scholar 

  37. Cooper C, Harvey NC, Dennison EM, van Staa TP. Update on the epidemiology of Paget’s disease of bone. J Bone Miner Res. 2006;21:3–8.

    Article  Google Scholar 

  38. Corral-Gudino L, Borao-Cengotita-Bengoa M, Del Pino-Monter J, Ralston SH. Epidemiology of Paget’s disease of bone: a systematic review and meta-analysis of secular changes. Bone. 2013;55:347–52.

    Article  PubMed  Google Scholar 

  39. Sankaran S, Naot D, Grey A, Cundy T. Paget’s disease in patients of Asian descent in New Zealand. J Bone Miner Res. 2012;27(1):223–6.

    Article  PubMed  Google Scholar 

  40. Ralston SH, Layfield R. Pathogenesis of Paget disease of bone. Calcif Tissue Int. 2012;91:97–113.

    Article  CAS  PubMed  Google Scholar 

  41. Bolland MJ, Cundy T. Paget’s disease of bone: clinical review and update. J Clin Pathol. 2013;66:924–7.

    Article  PubMed  Google Scholar 

  42. Goode A, Long JE, Shaw B, Ralston SH, Rios Visconti M, Gianfrancesco F, et al. Paget disease of bone-associated UBA domain mutations of SQSTM1 exert distinct effects on protein structure. Biochim Biophys Acta. 1842;2014:992–1000.

    Google Scholar 

  43. Chung PYJ, Van Hul W. Paget’s disease of bone: evidence for complex pathogenetic interactions. Semin Arth Rheum; 41:619–41.

  44. Daroszewska A, van’t Hof RJ, Rojas JA, Layfield R, Landao-Basonga E, Rose L, et al. A point mutation in the ubiquitin-associated domain of SQSMT1 is sufficient to cause a Paget’s disease like disorder in mice. Hum Mol Genet. 2011;20(14):2734–44.

    Article  CAS  PubMed  Google Scholar 

  45. Albagha OME, Rios Visconti M, Alonso N, Wani S, Goodman K, Fraser WD, et al. Common susceptibility alleles and SQSTM1 mutations predict disease extent and severity in a multinational study of patients with Paget’s disease. J Bone Miner Res. 2013;28(11):2338–46.

    Article  CAS  PubMed  Google Scholar 

  46. Rios Visconti M, Langston AL, Alonso N, Goodman K, Selby PL, Fraser WD, et al. Mutations of SQSTM1 are associated with severity and clinical outcome in Paget’s disease of bone. J Bone Miner Res. 2010;25(11):2368–73.

    Article  Google Scholar 

  47. Mills BG, Singer FR. Nuclear inclusions in Paget’s disease of bone. Science. 1976;194:201–2.

    Article  CAS  PubMed  Google Scholar 

  48. Deyrup AT, Montag AG, Inwards CY, Xu Z, Swee RG, Unni KK. Sarcomas arising in Paget disease of bone. A clinicopathologic analysis of 70 cases. Arch Pathol Lab Med. 2007;131:942–6.

    PubMed  Google Scholar 

  49. Fletcher CDM, Bridge JA, Hogendoorn PCW, Mertens F. WHO classification of tumours of soft tissue and bone. Pathology and genetics of tumours of soft tissue and bone. 4th ed. Lyon: IARC Press; 2013.

    Google Scholar 

Download references

Conflict of interest

The authors declare that they have no conflicts of interest.

Author information

Authors and Affiliations

  1. Department of Radiology, Royal Prince Alfred Hospital, PO Box M118, Missenden Road, Camperdown, NSW, 2050, Australia

    Salman Ansari & Wendy Brown

  2. Douglass Hanly Moir Pathology, 14 Giffnock Avenue, Macquarie Park, NSW, 2113, Australia

    Fiona Bonar

  3. Department of Orthopaedics, Royal Prince Alfred Hospital, Missenden Road, Camperdown, NSW, 2050, Australia

    Paul Stalley

Authors
  1. Salman Ansari
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Fiona Bonar
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Paul Stalley
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. Wendy Brown
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to Wendy Brown.

Rights and permissions

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Ansari, S., Bonar, F., Stalley, P. et al. Paget’s sarcoma of the patella. Skeletal Radiol 44, 1057–1063 (2015). https://doi.org/10.1007/s00256-015-2144-2

Download citation

  • Received:

  • Revised:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00256-015-2144-2

Keywords

Search

Navigation