Skip to main content
SpringerLink
Log in

Case Report: Unicameral Bone Cysts in a Young Patient with Acquired Generalized Lipodystrophy

  • Case Report
  • Published:
Clinical Orthopaedics and Related Research®

An Erratum to this article was published on 04 May 2010

Abstract

We report the case of a 13-year-old boy with bilateral distal femoral unicameral bone cysts (UBCs) associated with acquired generalized lipodystrophy. As opposed to congenital generalized lipodystrophy, cystic bone lesions in acquired generalized lipodystrophy are rare. After radiographic and histologic confirmation of the UBCs, we performed percutaneous intramedullary decompression, curettage, and grafting. UBCs can be an important manifestation of acquired generalized lipodystrophy. Cystic bone lesions appear to be less common in acquired generalized lipodystrophy than in congenital generalized lipodystrophy, and intramedullary adipose tissue loss may be a predisposing factor for the development of bone lesions in patients with acquired generalized lipodystrophy. When evaluating a patient with lipodystrophy, doctors should recognize the clinical course may include the development of UBCs.

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. 1A–B
Fig. 2A–B
Fig. 3
Fig. 4A–B
Fig. 5A–B
Fig. 6A–B
Fig. 7A–B
Fig. 8A–B

References

  1. Agarwal AK, Simha V, Oral EA, Moran SA, Gorden P, O’Rahilly S, Zaidi Z, Gurakan F, Arslanian SA, Klar A, Ricker A, White NH, Bindl L, Herbst K, Kennel K, Patel SB, Al-Gazali L, Garg A. Phenotypic and genetic heterogeneity in congenital generalized lipodystrophy. J Clin Endocrinol Metab. 2003;88:4840–4847.

    Article  CAS  PubMed  Google Scholar 

  2. Baig R, Eady JL. Unicameral (simple) bone cysts. South Med J. 2006;99:966–976.

    Article  PubMed  Google Scholar 

  3. Campanacci M, Capanna R, Picci P. Unicameral and aneurysmal bone cysts. Clin Orthop Relat Res. 1986;204:25–36.

    PubMed  Google Scholar 

  4. Chandalia M, Garg A, Vuitch F, Nizzi F. Postmortem findings in congenital generalized lipodystrophy. J Clin Endocrinol Metab. 1995;80:3077–3081.

    Article  CAS  PubMed  Google Scholar 

  5. Chigira M, Maehara S, Arita S, Udagawa E. The aetiology and treatment of simple bone cysts. J Bone Joint Surg Br. 1983;65:633–637.

    CAS  PubMed  Google Scholar 

  6. Coelho PC, Jordao A, Andre O, de Queiroz MV, Eurico-Lisboa P. Rheumatological manifestations of lipoatrophic diabetes. Clin Rheumatol. 1995;14:229–230.

    Article  CAS  PubMed  Google Scholar 

  7. Cohen J. Simple bone cysts: studies of cyst fluid in six cases with a theory of pathogenesis. J Bone Joint Surg Am. 1960;42:609–616.

    PubMed  Google Scholar 

  8. de Wazieres B, Wendling D, Test T, Morin G, Dupond JL. [Bone and visceral manifestations of lipoatrophic diabetes: apropos of a case] [in French]. Rev Rheum Mal Osteoartic. 1992;59:761–764.

    Google Scholar 

  9. Dormans JP, Sankar WN, Moroz L, Erol B. Percutaneous intramedullary decompression, curettage, and grafting with medical-grade calcium sulfate pellets for unicameral bone cysts in children: a new minimally invasive technique. J Pediatr Orthop. 2005;25:804–811.

    Article  PubMed  Google Scholar 

  10. Fleckenstein JL, Garg A, Bonte FJ, Vuitch MF, Peshock RM. The skeleton in congenital generalized lipodystrophy: evaluation using whole-body radiographic surveys, magnetic resonance imaging and technetium-99 m bone scintigraphy. Skeletal Radiol. 1992;21:381–386.

    Article  CAS  PubMed  Google Scholar 

  11. Garg A. Lipodystrophies. Am J Med. 2000;108:143–152.

    Article  CAS  PubMed  Google Scholar 

  12. Garg A, Peshock RM, Fleckenstein JL. Adipose tissue distribution pattern in patients with familial partial lipodystrophy (Dunnigan variety). J Clin Endocrinol Metab. 1999;84:170–174.

    Article  CAS  PubMed  Google Scholar 

  13. Goebel FD, Dorfler H, Goebel HH, Zollner N. Muscle capillary basement membrane thickness in lipoatrophic diabetes. Res Exp Med (Berl). 1977;171:271–276.

    Article  CAS  Google Scholar 

  14. Inoue O, Ibaraki K, Shimabukuro H, Shingaki Y. Packing with high-porosity hydroxyapatite cubes alone for the treatment of simple bone cyst. Clin Orthop Relat Res. 1993;293:287–292.

    PubMed  Google Scholar 

  15. Lodwick GS, Wilson AJ, Farrell C, Virtama P, Smeltzer FM, Dittrich F. Determining growth rates of focal lesions of bone from radiographs. Radiology. 1980;134:577–583.

    CAS  PubMed  Google Scholar 

  16. Mik G, Arkader A, Manteghi A, Dormans JP. Results of a minimally invasive technique for treatment of unicameral bone cysts. Clin Orthop Relat Res. 2009;467:2949–2954.

    Article  PubMed  Google Scholar 

  17. Misra A, Garg A. Clinical features and metabolic derangements in acquired generalized lipodystrophy: case reports and review of the literature. Medicine (Baltimore). 2003;82:129–146.

    Article  Google Scholar 

  18. Westvik J. Radiological features in generalized lipodystrophy. Acta Paediatr Suppl. 1996;413:44–51.

    Article  CAS  PubMed  Google Scholar 

  19. Wilson DE, Chan IF, Stevenson KB, Horton SC, Schipke C. Eucaloric substitution of medium chain triglycerides for dietary long chain fatty acids in acquired total lipodystrophy: effects on hyperlipoproteinemia and endogenous insulin resistance. J Clin Endocrinol Metab. 1983;57:517–523.

    Article  CAS  PubMed  Google Scholar 

Download references

Acknowledgments

We thank The Children’s Hospital of Philadelphia and the clinical research coordinators in the Department of Orthopaedics for their hard work.

Author information

Authors and Affiliations

  1. Department of Orthopaedic Surgery, Rush University, Chicago, IL, USA

    James M. Gregory MD

  2. Children’s Orthopaedic Center, Children’s Hospital Los Angeles, Keck School of Medicine, University of Southern California, Los Angeles, CA, USA

    Alexandre Arkader MD

  3. Department of Pathology, The Children’s Hospital of Philadelphia, Philadelphia, PA, USA

    Aqiba Bothari MD

  4. Department of Orthopaedic Surgery, The Children’s Hospital of Philadelphia, University of Pennsylvania School of Medicine, 2nd Floor Wood Center, 34th Street and Civic Center Boulevard, Philadelphia, PA, 19104-4399, USA

    John P. Dormans MD

Authors
  1. James M. Gregory MD
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Alexandre Arkader MD
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Aqiba Bothari MD
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. John P. Dormans MD
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to John P. Dormans MD.

Additional information

Each author certifies that he or she has no commercial associations (eg, consultancies, stock ownership, equity interest, patent/licensing arrangements, etc) that might pose a conflict of interest in connection with the submitted article.

Each author certifies that his or her institution has approved the reporting of this case, that all the investigations were conducted in conformity with ethical principles of research, and that informed consent for participation in the study was obtained.

This work was performed at The Children’s Hospital of Philadelphia.

An erratum to this article can be found at http://dx.doi.org/10.1007/s11999-010-1371-z

About this article

Cite this article

Gregory, J.M., Arkader, A., Bothari, A. et al. Case Report: Unicameral Bone Cysts in a Young Patient with Acquired Generalized Lipodystrophy. Clin Orthop Relat Res 468, 1440–1446 (2010). https://doi.org/10.1007/s11999-009-1168-0

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s11999-009-1168-0

Keywords

Search

Navigation