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Bone histomorphometry in a long-term hemodialysis patient with hypoparathyroidism and sarcoidosis

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Abstract

A bone biopsy specimen in a long-term hemodialysis patient with sarcoidosis coexisting with severe hypoparathyroidism has demonstrated that a persistent near physiological level of 1,25-dihydroxyvitamin D3 contributes to the preservation of bone remodeling and has the potential to retard the development of vascular calcification and atherosclerosis. Sarcoidosis-related hypercalcemia and hypoparathyroidism, which is characterized by 1,25-dihydroxyvitamin D3 (1,25(OH)2D3) overproduction, is rarely seen in hemodialysis patients. Herein, we describe a 60-year-old Japanese woman on hemodialysis for 35 years who presented with malaise and hypercalcemia. Severe hypoparathyroidism without parathyroidectomy and a preserved 1,25(OH)2D3 level were detected. Computed tomography showed bilateral axillary lymphadenopathy and minimal aortic and soft tissue calcification. The axillary node biopsy led to a definite diagnosis of sarcoidosis. A bone biopsy specimen obtained from the right iliac crest showed remodeling of normal lamellar bone with scalloped cement lines and clear double labeling by tetracycline on fluorescence microscopy. Histomorphometric analysis revealed that the bone formation rate was preserved (30.0 %/year), together with a decrease of osteoid volume (5.75 %) and fibrous volume (0 %), indicating that the patient did not have adynamic bone disease and only showed mild disease. This is the first documented case of sarcoidosis-related hypercalcemia associated with severe hypoparathyroidism in a long-term hemodialysis patient who underwent bone histomorphometry. Our findings suggest that, in hemodialysis patients with sarcoidosis coexisting with severe hypoparathyroidism, a persistent near physiological level of 1,25(OH)2D3 contributes to the preservation of bone remodeling and has the potential to retard the development of vascular calcification and atherosclerosis.

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References

  1. Iannuzzi MC, Rybicki BA, Teirstein AS (2007) Sarcoidosis. N Engl J Med 357:2153–2165

    Article  CAS  PubMed  Google Scholar 

  2. Taylor RL, Lynch HJ Jr, Wysor WG Jr (1963) Seasonal influence of sunlight on the hypercalcemia of sarcoidosis. Am J Med 34:221–227

    Article  CAS  PubMed  Google Scholar 

  3. Kitahara T, Ono K, Tsuchida A, Kawai H, Shinohara M, Ishii Y, Koyanagi H, Noguchi T, Matsumoto T, Sekihara T, Watanabe Y, Kanai H, Ishida H, Nojima Y (2005) Impact of brachial-ankle pulse wave velocity and ankle-brachial blood pressure index on mortality in hemodialysis patients. Am J Kidney Dis 46:688–696

    Article  PubMed  Google Scholar 

  4. Nishizawa Y, Shoji T, Maekawa K, Nagasue K, Okuno S, Kim M, Emoto M, Ishimura E, Nakatani T, Miki T, Inaba M (2003) Intima-media thickness of carotid artery predicts cardiovascular mortality in hemodialysis patients. Am J Kidney Dis 41:S76–S79

    Article  PubMed  Google Scholar 

  5. Tanizawa T, Itoh A, Uchiyama T, Zhang L, Yamamoto N (1999) Changes in cortical width with bone turnover in the three different endosteal envelopes of the ilium in postmenopausal osteoporosis. Bone 25:493–499

    Article  CAS  PubMed  Google Scholar 

  6. Parfitt AM, Drezner MK, Glorieux FH, Kanis JA, Malluche H, Meunier PJ, Ott SM, Recker RR (1987) Bone histomorphometry: standardization of nomenclature, symbols, and units. Report of the ASBMR Histomorphometry Nomenclature Committee. J Bone Miner Res 2:595–610

    Article  CAS  PubMed  Google Scholar 

  7. Rocker RR, Kimmel DB, Parfitt AM, Davies KM, Keshawarz N, Hinders S (1988) Static and tetracyclin-based bone histomorphometric data from 34 normal postmenopausal females. J Bone Miner Res 3:133–144

    Article  Google Scholar 

  8. Kobayashi S, Takahashi HE, Ito A, Saito N, Nawata M, Horiuchi H, Ohta H, Iorio R, Yamamoto N, Takaoka K (2003) Trabecular minimodeling in human iliac bone. Bone 32:163–169

    Article  CAS  PubMed  Google Scholar 

  9. Sherrard DJ, Hercz G, Pei Y, Maloney NA, Greenwood C, Manuel A, Saiphoo C, Fenton SS, Segre GV (1993) The spectrum of bone disease in end-stage renal failure—an evolving disorder. Kidney Int 43:436–442

    Article  CAS  PubMed  Google Scholar 

  10. Barbour GL, Coburn JW, Slatopolsky E, Norman AW, Horst RL (1981) Hypercalcemia in an anephric patient with sarcoidosis: evidence for external generation of 1,25-dihydroxyvitamin D. N Engl J Med 305:440–443

    Article  CAS  PubMed  Google Scholar 

  11. Kalantar-Zadeh K, Neumayer HH, Wunsch PH, Luft FC (1994) Hypercalcaemia and sarcoidosis in an anephric dialysis patient. Nephrol Dial Transplant 9:829–831

    CAS  PubMed  Google Scholar 

  12. Hrrero JC, Morales E, Dominguez-Gil B, Carreno A, Usera G, Aguado JM, Praga M (1998) Reactivation of multisystemic sarcoidosis after immunosuppression withdrawal in a transplanted patient returning to chronic dialysis. Nephrol Dial Transplant 13:3280–3281

    Article  Google Scholar 

  13. Naito T (1999) Progressive tumoral calcinosis as the presenting feature of sarcoidosis in a patient on haemodialysis treatment. Nephrol Dial Transplant 14:2716–2719

    Article  CAS  PubMed  Google Scholar 

  14. Barnard S, Marshall R, Parry RG, Johnston P (2002) Sarcoid presenting with hypercalcemia and erythema nodosum in a haemodialysis patient. Nephrol Dial Transplant 17:175

    Article  PubMed  Google Scholar 

  15. Sato T, Tsuru T, Hagiwara K, Miyashita K, Matsuno H, Goto A, Oritsu M, Hamanaka T, Akiyama O (2006) Sarcoidosis with acute recurrent polyarthritis and hypercalcemia. Intern Med 45:363–368

    Article  PubMed  Google Scholar 

  16. Huart A, Kamar N, Lanau JM, Dahmani A, Durand D, Rostaing L (2006) Sarcoidosis-related hypercalcemia in 3 chronic hemodialysis patients. Clin Nephrol 65:449–452

    Article  CAS  PubMed  Google Scholar 

  17. Quack I, Woznowski M, Schieren G, Weiner SM, Winnekendonk G, Tokmak F, Rump LC, Rattensperger D (2007) Hypercalcemia after transplant nephrectomy in a hemodialysis patient: a case report. J Med Case Rep 3:164

    Article  Google Scholar 

  18. Yamada S, Taniguchi M, Tsuruya K, Iida M (2009) Recurrent sarcoidosis with psoas muscle granuloma and hypercalcaemia in a patient on chronic haemodialysis. Nephrology (Carlton) 14:452–453

    Article  Google Scholar 

  19. Horikoshi R, Akimoto T, Meguro D, Saito O, Ando Y, Muto S, Kusano E (2011) Tumoral calcinosis associated with hypercalcemia in a patient with chronic renal failure. Clin Exp Nephrol 15:154–158

    Article  PubMed  Google Scholar 

  20. Kwon A, Koh ES, Chung S, Kim YK (2013) Sarcoidosis as a cause of unappreciated hypercalcaemia in a patient with end-stage renal disease on peritoneal dialysis. BMJ Case Rep. doi:10.1136/bcr-2013-010017

    Google Scholar 

  21. Arai Y, Tanaka H, Hirasawa S, Aki S, Inaba N, Aoyagi M, Tsuura Y, Tamura T (2013) Sarcoidosis in a chronic dialysis patient diagnosed by sarcoidosis-related hypercalcemia with no common systemic clinical manifestations: a case report and review of the literature. Intern Med 52:2639–2644

    Article  PubMed  Google Scholar 

  22. Brandi L, Daugaard H, Tvedegaard E, Storm T, Olgaard K (1989) Effect of intravenous 1-alpha-hydroxyvitamin D3 on secondary hyperparathyroidism in chronic uremic patients on maintenance hemodialysis. Nephron 53:194–200

    Article  CAS  PubMed  Google Scholar 

  23. Baker LR, Abrams L, Roe CJ, Faugere MC, Fanti P, Subayti Y, Malluche HH (1989) 1,25(OH)2D3 administration in moderate renal failure: a prospective double-blind trial. Kidney Int 35:661–669

    Article  CAS  PubMed  Google Scholar 

  24. Gallieni M, Kamimura S, Ahmed A, Bravo E, Delmez J, Slatopolsky E, Dusso A (1995) Kinetics of monocyte 1 alpha-hydroxylase in renal failure. Am J Physiol 268:F746–F753

    CAS  PubMed  Google Scholar 

  25. Couttenye MM, D’Haese PC, Verschoren WJ, Behets GJ, Schrooten I, De Broe ME (1999) Low bone turnover in patients with renal failure. Kidney Int 73:S70–S76

    Article  CAS  Google Scholar 

  26. London GM, Marchais SJ, Guerin AP, Boutouyrie P, Metivier F, de Vernejoul MC (2008) Association of bone activity, calcium load, aortic stiffness, and calcifications in ESRD. J Am Soc Nephrol 19:1827–1835

    Article  PubMed Central  CAS  PubMed  Google Scholar 

  27. Gerakis A, Hutchison AJ, Apostolou T, Freemont AJ, Billis A (1996) Biochemical markers for non-invasive diagnosis of hyperparathyroid bone disease and adynamic bone in patients on haemodialysis. Nephrol Dial Transplant 11:2430–2438

    Article  CAS  PubMed  Google Scholar 

  28. Qi Q, Monier-Faugere MC, Geng Z, Malluche HH (1995) Predictive value of serum parathyroid hormone levels for bone turnover in patients on chronic maintenance dialysis. Am J Kidney Dis 26:622–631

    Article  CAS  PubMed  Google Scholar 

  29. Ubara Y, Tagami T, Nakanishi S, Sawa N, Hoshino J, Suwabe T, Katori H, Takemoto F, Hara S, Takaichi K (2005) Significance of minimodeling in dialysis patients with adynamic bone disease. Kidney Int 68:833–839

    Article  PubMed  Google Scholar 

  30. Sutton AL, Zhang X, Ellison TI, Macdonald PN (2005) The 1,25(OH)2D3-regulated transcription factor MN1 stimulates vitamin D receptor-mediated transcription and inhibits osteoblastic cell proliferation. Mol Endocrinol 19:2234–2244

    Article  CAS  PubMed  Google Scholar 

  31. van Leeuwen JP, van Driel M, van den Bemd GJ, Pols HA (2001) Vitamin D control of osteoblast function and bone extracellular matrix mineralization. Crit Rev Eukaryot Gene Expr 11:199–226

    Article  PubMed  Google Scholar 

  32. London GM (2004) Arterial calcifications and bone histomorphometry in end-stage renal disease. J Am Soc Nephrol 15:1943–1951

    Article  PubMed  Google Scholar 

  33. Goldsmith DJ, Covic A, Sambrook PA, Ackrill P (1997) Vascular calcification in long-term haemodialysis patients in a single unit: a retrospective analysis. Nephron 77:37–43

    Article  CAS  PubMed  Google Scholar 

  34. Milliner DS, Zinsmeister AR, Lieberman E, Landing B (1990) Soft tissue calcification in pediatric patients with end-stage renal disease. Kidney Int 38:931–936

    Article  CAS  PubMed  Google Scholar 

  35. Levin A, Li YC (2005) Vitamin D and its analogues: do they protect against cardiovascular disease in patients with kidney disease? Kidney Int 68:1973–1981

    Article  CAS  PubMed  Google Scholar 

  36. Naves-Diaz M, Alvarez-Hernandez D, Passlick-Deetjen J, Guinsburg A, Marelli C, Rodriguez-Puyol D, Cannata-Andia JB (2008) Oral active vitamin D is associated with improved survival in hemodialysis patients. Kidney Int 74:1070–1078

    Article  CAS  PubMed  Google Scholar 

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Acknowledgments

This study was partially supported by the Okinaka Memorial Institute for Medical Research. We wish to acknowledge Akemi Ito (Ito Bone Histomorphometry Institute) for performing the bone histomorphometric analysis.

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Correspondence to K. Sumida.

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Sumida, K., Ubara, Y., Hoshino, J. et al. Bone histomorphometry in a long-term hemodialysis patient with hypoparathyroidism and sarcoidosis. Osteoporos Int 26, 1435–1441 (2015). https://doi.org/10.1007/s00198-014-2987-8

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  • DOI: https://doi.org/10.1007/s00198-014-2987-8

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