Skip to main content

Bone Histomorphometry in Miscellaneous Metabolic Diseases: Hepatic C-Associated Osteosclerosis, IgG4-Related Disease, and Ehlers–Danlos Syndrome

  • Chapter
  • First Online:
Osteoporotic Fracture and Systemic Skeletal Disorders

Abstract

Quantitative analysis by bone histomorphometry provides information about bone quality and dynamic bone turnover. In this chapter we present histomorphometric analysis of three specific systemic diseases with skeletal disorders. Histomorphometric assessments in hepatic C-associated osteosclerosis, IgG4-related disease, and Ehlers–Danlos syndrome were performed in these cases to clarify bone metabolism and structure. All these cases revealed unknown histomorphometric features in bone biopsies. Hepatic C-associated osteosclerosis demonstrated a thicker cortex with excessive bone formation, IgG4-related disease revealed unusual demineralization of bone, and Ehlers–Danlos syndrome indicated low turnover bone.

The present invited review was completed and submitted to the publisher on 30-May-20. 

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

Access this chapter

Chapter
USD 29.95
Price excludes VAT (USA)
  • Available as PDF
  • Read on any device
  • Instant download
  • Own it forever
eBook
USD 84.99
Price excludes VAT (USA)
  • Available as EPUB and PDF
  • Read on any device
  • Instant download
  • Own it forever
Softcover Book
USD 109.99
Price excludes VAT (USA)
  • Compact, lightweight edition
  • Dispatched in 3 to 5 business days
  • Free shipping worldwide - see info
Hardcover Book
USD 159.99
Price excludes VAT (USA)
  • Durable hardcover edition
  • Dispatched in 3 to 5 business days
  • Free shipping worldwide - see info

Tax calculation will be finalised at checkout

Purchases are for personal use only

Institutional subscriptions

References

  1. Takahashi H, et al. Evidence that bone resorption proceeds formation at cellular level. Henry Ford Hosp Med Bull. 1964;12:359–64.

    Google Scholar 

  2. Parfitt AM, et al. Bone histomorphometry: standardization of nomenclature, symbols, and units. Report of the ASBMR Histomorphometry Nomenclature Committee. J Bone Miner Res. 1987;2:595–610.

    Article  CAS  Google Scholar 

  3. Dempster DW, et al. Standardized nomenclature, symbols, and units for bone histomorphometry: a 2012 update of the report of the ASBMR Histomorphometry Nomenclature Committee. J Bone Miner Res. 2013;28:2–17.

    Google Scholar 

  4. Chavassieux P, et al. Clinical use of bone biopsy. In: Marcus R, editor. Osteoporosis. 2nd ed. Academic; 2001. p. 501–9.

    Chapter  Google Scholar 

  5. Recker RR. Bone biopsy and histomorphometry in clinical practice, 7th ed. Primer on the metabolic bone disease and disorders of mineral metabolism, ASBMR; 2008. p. 180–6.

    Google Scholar 

  6. Recker RR, et al. Issues in modern bone histomorphometry. Bone. 2011;49:955–64.

    Article  CAS  Google Scholar 

  7. Yamamoto N, et al. Bone cell biology assessed by microscopic approach. Bone histomorphometry of remodeling, modeling and minimodeling. Clin Calcium. 2015;25:1491–7.

    CAS  PubMed  Google Scholar 

  8. Miyamura N, et al. A case of hepatitis C-associated osteosclerosis: accelerated bone turnover controlled by pulse steroid therapy. Endocri Diabet Metab. 2016.

    Google Scholar 

  9. Beighton P, et al. A review of the osteoscleroses. Postgrad Med J. 1977;53:507.

    Article  CAS  Google Scholar 

  10. Whyte MP. Heritable metabolic and dysplastic bone disease. Endocrinol Metab Clin N Am. 1990;19:133.

    Article  CAS  Google Scholar 

  11. Villareal DT, et al. Painful diffuse osteosclerosis after intravenous drug abuse. Am J Med. 1992;93:371–81.

    Article  CAS  Google Scholar 

  12. Serraino C, et al. Hepatic C-associated osteosclerosis: a review of the literature. Intern Med. 2015;54:777–83.

    Article  CAS  Google Scholar 

  13. Epperle N, et al. An uncommon cause of acquired osteosclerosis in adults: hepatitis C-associated osteosclerosis. Skeletal Radiol. 2014;43:1313–8.

    Article  Google Scholar 

  14. Khosla S, et al. Use of site-specific antibodies to characterize the circulating form of big insulin-like growth factor II in patients with hepatitis C-associated osteosclerosis. J Clin Endocrinol Metab. 2002;87:3867–70.

    Article  CAS  Google Scholar 

  15. Manganelli, et al. POPG/RANKL system imbalance in a case of hepatitis C-associated osteosclerosis: the pathogenetic key? 2005;24:296–300.

    Google Scholar 

  16. Kaji H, et al. Serum soluble factors induce the proliferation, alkaline phosphatase activity and transforming growth factor-beta signal in osteoblastic cells in the patient with hepatitis C-associated osteosclerosis. Exp Clin Endocrinol Diabetes. 2006;114:599–604.

    Article  CAS  Google Scholar 

  17. Javier R, et al. Skeletal recovery from hepatitis C-associated osteosclerosis (HCAO) following antiviral treatment. Joint Bone Spine. 2011;78:409–11.

    Article  Google Scholar 

  18. Hassoum AA, et al. Hepatic C-associated osteosclerosis: an unusual syndrome of acquired osteosclerosis in adults. Am J Med. 1997;103:70–3.

    Article  Google Scholar 

  19. Kawakita C, et al. A case of fatal osteolytic hypercalcemia complicated with IgG4-related ophthalmic disease leading to renal failure. CEN Case Rep. 2019;8:23–30.

    Google Scholar 

  20. Bilezikian JP. Management of acute hypercalcemia. N Engl J Med. 1992;326:1196–203.

    Article  CAS  Google Scholar 

  21. Umehara H, et al. A novel clinical entity, IgG4-related disease: general concept and details. Med Rheumatol. 2012;22:1–14.

    Article  CAS  Google Scholar 

  22. Stone JH, et al. IgG4 disease. N Engl J Med. 2012;366:539–51.

    Article  CAS  Google Scholar 

  23. Ichiki A, et al. IgG4-related disease with bone marrow involvement. Int Med. 2016;55:2295–9.

    Article  CAS  Google Scholar 

  24. Uehara M, et al. Spinal manifestations in 12 patients with musculocontractural Ehlers-Danlos syndrome caused by CHST14/D4ST1 deficiency (mcEDS-CHST14). Am L Med Genet. 2018;176:2331–41.

    Article  CAS  Google Scholar 

  25. Steinmann B, Royce PM, Superti-Furga A. The Ehlers-Danlos syndrome. In: Royce PM, Steinmann B, editors. Connective tissue and its heritable disorders. New York: Wiley-Liss; 2002. p. 431–523.

    Chapter  Google Scholar 

  26. Jasiewicz B, Potaczek T, Tesiorowski M, Lokas K. Spine deformities in patients with Ehlers-Danlos syndrome, type IV – late results of surgical treatment. Scoliosis. 2010;5:26.

    Article  Google Scholar 

  27. Akpinar S, et al. Surgical management of the spinal deformity in Ehlers-Danlos syndrome type VI. Eur Spine J. 12;135–140.

    Google Scholar 

  28. Liu Y, et al. Posterior spinal fusion for scoliosis in Ehlers-Danlos syndrome, kyphoscoliosis type. Orthopedics. 2011;34:228.

    PubMed  Google Scholar 

  29. Mazziotti G, et al. High prevalence of radiological vertebral fractures in adult patients with Ehlers-Danlos syndrome. Bone. 2016;84:88–92.

    Google Scholar 

  30. Eller-Vainicher C, et al. Bone involvement in adult patients affected with Ehlers-Danlos syndrome. Osteoporos Int. 2016;27:2525–31.

    Article  CAS  Google Scholar 

Download references

Author information

Authors and Affiliations

Authors

Corresponding author

Correspondence to Noriaki Yamamoto .

Editor information

Editors and Affiliations

Rights and permissions

Reprints and permissions

Copyright information

© 2022 Springer Nature Singapore Pte Ltd.

About this chapter

Check for updates. Verify currency and authenticity via CrossMark

Cite this chapter

Yamamoto, N. et al. (2022). Bone Histomorphometry in Miscellaneous Metabolic Diseases: Hepatic C-Associated Osteosclerosis, IgG4-Related Disease, and Ehlers–Danlos Syndrome. In: Takahashi, H.E., Burr, D.B., Yamamoto, N. (eds) Osteoporotic Fracture and Systemic Skeletal Disorders. Springer, Singapore. https://doi.org/10.1007/978-981-16-5613-2_20

Download citation

  • DOI: https://doi.org/10.1007/978-981-16-5613-2_20

  • Published:

  • Publisher Name: Springer, Singapore

  • Print ISBN: 978-981-16-5612-5

  • Online ISBN: 978-981-16-5613-2

  • eBook Packages: MedicineMedicine (R0)

Publish with us

Policies and ethics