Calcified Tissue International

, Volume 102, Issue 3, pp 368–372 | Cite as

Multiple Vertebral Osteonecroses (Kümmell’s Disease) After 10 Years on Denosumab: Is Osteocyte Apoptosis to Blame?

  • Yves Maugars
  • Géraldine Bart
  • Pascale Guillot
  • Marguerite Chemel-Mary
  • Joëlle Glémarec
  • Mélanie Gahier-Penhoat
  • Benoit Le Goff
  • Christelle Darrieutort-Laffite
Case Reports


We report here a case of multiple vertebral osteonecroses with intrasomatic gaseous dissection (Kümmell’s disease) occurring 1 year after the end of a 10-year course of denosumab treatment for osteoporosis without fractures. Histomorphometry and bone remodeling markers revealed major bone resorption and the persistence of an inhibition of bone formation. The presence of multiple empty lacunae in the bone provided evidence for high levels of osteocyte apoptosis. Osteocytes direct bone resorption (via the RANK/RANK-L/osteoprotegerin system) and formation (Wnt system, with SOST and DKK1) pathways. The vertebral osteonecrosis in our case may, therefore, have resulted from osteocyte apoptosis, decompensated by the sudden reactivation of bone remodeling after the cessation of denosumab treatment.


Osteoporosis Vertebral osteonecrosis Denosumab Osteocyte apoptosis 



The authors would like to thank Pr Daniel Chappard, MD PD, Department of cellular and tissular pathology, University of Angers, France.

Compliance with ethical standards

Conflict of interest

Yves Maugars, Géraldine Bart, Pascale Guillot, Marguerite Chemel-Mary, Joëlle Glémarec, Mélanie Gahier-Penhoat, Benoit Le Goff, and Christelle Darrieutort-Laffite declare no conflict of interest.

Human and Animal Rights and Informed Consent

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.


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Copyright information

© Springer Science+Business Media, LLC 2017

Authors and Affiliations

  • Yves Maugars
    • 1
  • Géraldine Bart
    • 1
  • Pascale Guillot
    • 1
  • Marguerite Chemel-Mary
    • 1
  • Joëlle Glémarec
    • 1
  • Mélanie Gahier-Penhoat
    • 1
  • Benoit Le Goff
    • 1
  • Christelle Darrieutort-Laffite
    • 1
  1. 1.Rheumatology DepartmentNantes University HospitalNantes CedexFrance

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