Skip to main content
Log in

Looking beyond low bone mineral density: Multiple insufficiency fractures in a woman with post-menopausal osteoporosis on alendronate therapy

  • Case Report
  • Published:
Journal of Endocrinological Investigation Aims and scope Submit manuscript

Abstract

Insufficiency fractures occur most commonly in the pelvic girdle and in the sacrum, followed by the tibia and the femoral neck. Insufficiency fractures of the femoral diaphyses are rare, with only few reported cases in the literature. The strongest associations exist with untreated osteoporosis. We describe an unusual case of multiple insufficiency fractures in a 73-yr-old Chinese woman who presented with a 10-month history of bilateral groin pain and difficulty with walking in the absence of trauma, diagnosed 18 months following the commencement of anti-resorptive therapy with alendronate. The pathogenesis of such insufficiency fractures is poorly understood, but next to low bone mineral density and micro-architectural damage likely involves other components such as changes in bone turnover and patient-related factors (e.g. non-compliance). This case report and review of the literature draws attention to some of the challenges in the diagnosis and management of such rare insufficiency fractures.

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

Access this article

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

Instant access to the full article PDF.

Similar content being viewed by others

References

  1. Kanis JA. Assessment of fracture risk and its application to screening for postmenopausal osteoporosis: synopsis of a WHO report. WHO Study Group. Osteoporos Int 1994, 4: 368–81.

    Article  CAS  Google Scholar 

  2. Melton LJ 3rd, Atkinson EJ, O’Fallon WM, Wahner HW, Riggs BL. Long-term fracture prediction by bone mineral assessed at different skeletal sites. J Bone Miner Res 1993, 8: 227–33.

    Google Scholar 

  3. Marshall D, Johnell O, Wedel H. Meta-analysis of how well measures of bone mineral density predict occurrence of osteoporotic fractures. BMJ 1996, 312: 1254–9.

    Article  PubMed Central  PubMed  CAS  Google Scholar 

  4. Marwick C. Consensus panel considers osteoporosis. JAMA 2000, 283: 2093–5.

    Article  PubMed  CAS  Google Scholar 

  5. Frost HM. A 2003 update of bone physiology and Wolff’s Law for clinicians. Angle Orthod 2004, 74: 3–15.

    PubMed  Google Scholar 

  6. Christiansen CG, Kassim RA, Callaghan JJ, Marsh JL, Schmidt AH. Pubic ramus insufficiency fractures following total hip arthroplasty. A report of six cases. J Bone Joint Surg Am 2003, 85: 1819–22.

    Google Scholar 

  7. Ubara Y, Higa Y, Tagami T, et al. Pelvic insufficiency fracture related to autosomal dominant polycystic kidney disease. Am J Kidney Dis 2005, 46: e103–11.

    Article  PubMed  Google Scholar 

  8. Inoue Y, Miki C, Ojima E, Nomoto Y, Kusunoki M. Pelvic insufficiency fractures after preoperative radiotherapy for rectal carcinoma. Int J Clin Oncol 2003, 8: 336–9.

    Article  PubMed  Google Scholar 

  9. Ogino I, Okamoto N, Ono Y, Kitamura T, Nakayama H. Pelvic insufficiency fractures in postmenopausal woman with advanced cervical cancer treated by radiotherapy. Radiother Oncol 2003, 68: 61–7.

    Article  PubMed  Google Scholar 

  10. Hoshino Y, Doita M, Yoshikawa M, Hirayama K, Sha N, Kurosaka M. Unstable pelvic insufficiency fracture in a patient with rheumatoid arthritis. Rheumatol Int 2004, 24: 46–9.

    Article  PubMed  Google Scholar 

  11. Huh SJ, Kim B, Kang MK, et al. Pelvic insufficiency fracture after pelvic irradiation in uterine cervix cancer. Gynecol Oncol 2002, 86: 264–8.

    Article  PubMed  Google Scholar 

  12. Chan K, Resnick D, Pathria M, Jacobson J. Pelvic instability after bone graft harvesting from posterior iliac crest: report of nine patients. Skeletal Radiol 2001, 30: 278–81.

    Article  PubMed  CAS  Google Scholar 

  13. Moreno A, Clémente J, Crespo C, et al. Pelvic insufficiency fractures in patients with pelvic irradiation. Int J Radiat Oncol Biol Phys 1999, 44: 61–6.

    Article  PubMed  CAS  Google Scholar 

  14. liner K, Blomlie V, Fosså SD. Radiation-induced insufficiency fractures after definitive prostate cancer radiotherapy. Acta Oncol 1998, 37: 201–2.

    Article  Google Scholar 

  15. Parikh VA, Edlund JW. Sacral insufficiency fractures—rare complication of pelvic radiation for rectal carcinoma: report of a case. Dis Colon Rectum 1998, 41: 254–7.

    Article  PubMed  CAS  Google Scholar 

  16. Rex C, Elsworth C. Pelvic insufficiency fracture with diastasis of the pubic symphysis after irradiation: a case report. J Bone Joint Surg Br 1998, 80: 264–6.

    Article  PubMed  CAS  Google Scholar 

  17. Mumber MP, Greven KM, Haygood TM. Pelvic insufficiency fractures associated with radiation atrophy: clinical recognition and diagnostic evaluation. Skeletal Radiol 1997, 26: 94–9.

    Article  PubMed  CAS  Google Scholar 

  18. Blomlie V, Rofstad EK, Talle K, Sundfor K, Winderen M, Lien HH. Incidence of radiation-induced insufficiency fractures of the female pelvis: evaluation with MR imaging. AJR Am J Roentgenol 1996, 167: 1205–10.

    Article  PubMed  CAS  Google Scholar 

  19. Burnet S, Mahadevan G, Lee A, Rischmueller M. Sacral insufficiency fracture—a case of post-coital back pain. Rheumatology (Oxford) 2001, 40: 1065–6.

    Article  CAS  Google Scholar 

  20. Khan MH, Smith PN, Kang JD. Sacral insufficiency fractures following multilevel instrumented spinal fusion: case report. Spine 2005, 30: 484–8.

    Article  Google Scholar 

  21. Wild A, Jaeger M, Haak H, Mehdian SH. Sacral insufficiency fracture, an unsuspected cause of low-back pain in elderly women. Arch Orthop Trauma Surg 2002, 122: 58–60.

    Article  PubMed  CAS  Google Scholar 

  22. Lin J, Lachmann E, Nagler W. Sacral insufficiency fractures: a report of two cases and a review of the literature. J Womens Health Gend Based Med 2001, 10: 699–705.

    Article  PubMed  CAS  Google Scholar 

  23. Schmid L, Pfirrmann C, Hess T, Schlumpf U. Bilateral fracture of the sacrum associated with pregnancy: a case report. Osteoporos Int 1999, 10: 91–3.

    Article  PubMed  CAS  Google Scholar 

  24. Schulman LL, Addesso V, Staron RB, McGregor CC, Shane E. Insufficiency fractures of the sacrum: a cause of low back pain after lung transplantation. J Heart Lung Transplant 1997, 16: 1081–5.

    PubMed  CAS  Google Scholar 

  25. Breuil V, Brocq O, Euller-Ziegler L, Grimaud A. Insufficiency fracture of the sacrum revealing a pregnancy associated osteoporosis. First case report. Ann Rheum Dis 1997, 56: 278–9.

    Article  CAS  Google Scholar 

  26. Clemetson IA, Popp A, Lippuner K, Ballmer F, Anderson SE. Postpartum osteoporosis associated with proximal tibial stress fracture. Skeletal Radiol 2004, 33: 96–8.

    Article  PubMed  CAS  Google Scholar 

  27. Franco M, Bendini JC, Blaimont A, Albano L, Cassuto E, Jaeger P. Longitudinal bone insufficiency fracture of the tibia in a renal transplant recipient. Joint Bone Spine 2003, 70: 296–9.

    Article  PubMed  Google Scholar 

  28. Vanhoof J, Landewe S, Vandevenne J, Geusens P. An exceptional radiographic presentation of bilateral insufficiency fractures of the proximal tibia in a patient with rheumatoid arthritis. Ann Rheum Dis 2003, 62: 277–9.

    Article  PubMed Central  PubMed  CAS  Google Scholar 

  29. Feydy A, Carlier R, Mutschler C, Bernard L, Leriverend V, Vallee C. A longitudinal insufficiency fracture of the tibia in association with a healed chronic osteomyelitis. Eur Radiol 2000, 10: 1929–31.

    Article  PubMed  CAS  Google Scholar 

  30. Daunt N, Gribbin D, Slater GS. Longitudinal tibial stress fractures. Australas Radiol 1998, 42: 188–90.

    Article  PubMed  CAS  Google Scholar 

  31. Ikemura S, Yamamoto T, Nakashima Y, Shuto T, Jingushi S, Iwamoto Y Bilateral subchondral insufficiency fracture of the femoral head after renal transplantation: a case report. Arthritis Rheum 2005, 52: 1293–6.

    Article  PubMed  Google Scholar 

  32. Huang KC, Hsu WH, Lee KF, Hsu RW. Subchondral insufficiency fracture with rapid collapse of the femoral head in a patient with Turner’s syndrome. Rheumatology (Oxford) 2005, 44: 826–7.

    Article  Google Scholar 

  33. Motomura G, Yamamoto T, Miyanishi K, Shirasawa K, Noguchi Y, Iwamoto Y. Subchondral insufficiency fracture of the femoral head and acetabulum: a case report. J Bone Joint Surg Am 2002, 84: 1205–9.

    Article  PubMed  Google Scholar 

  34. Yamamoto T, Takabatake K, Iwamoto Y. Subchondral insufficiency fracture of the femoral head resulting in rapid destruction of the hip joint: a sequential radiographic study. AJR Am J Roentgenol 2002, 178: 435–7.

    Article  PubMed  Google Scholar 

  35. Yamamoto T, Bullough PG. The role of subchondral insufficiency fracture in rapid destruction of the hip joint: a preliminary report. Arthritis Rheum 2000, 43: 2423–7.

    Article  PubMed  CAS  Google Scholar 

  36. Niimi R, Hasegawa M, Sudo A, Uchida A. Rapidly destructive coxopathy after subchondral insufficiency fracture of the femoral head. Arch Orthop Trauma Surg 2005, 125: 410–3.

    Article  PubMed  Google Scholar 

  37. Legroux Gerot I, Demondion X, Louville AB, Delcambre B, Cortet B. Subchondral fractures of the femoral head: a review of seven cases. Joint Bone Spine 2004, 71: 131–5.

    Article  PubMed  Google Scholar 

  38. Davies M, Cassar-Pullicino VN, Darby AJ. Subchondral insufficiency fractures of the femoral head. Eur Radiol 2004, 14: 201–7.

    Article  PubMed  CAS  Google Scholar 

  39. Yamamoto T, Schneider R, Bullough PG. Subchondral insufficiency fracture of the femoral head: histopathologic correlation with MRI. Skeletal Radiol 2001, 30: 247–54.

    Article  PubMed  CAS  Google Scholar 

  40. Yamamoto T, Schneider R, Bullough PG. Insufficiency subchondral fracture of the femoral head. Am J Surg Pathol 2000, 24: 464–8.

    Article  PubMed  CAS  Google Scholar 

  41. Kitajima I, Tachibana S, Mikami Y, Mori T, Hirota Y, Nakamichi K. Insufficiency fracture of the femoral neck after intramedullary nailing. J Orthop Sci 1999, 4: 304–6.

    Article  PubMed  CAS  Google Scholar 

  42. Egol KA, Koval KJ, Kummer F, Frankel VH. Stress fractures of the femoral neck. Clin Orthop Relat Res 1998, 348: 72–8.

    PubMed  Google Scholar 

  43. Kageyama Y, Nagafusa T, Nagano A. Insufficiency fracture of the tarsal navicular in a patient with rheumatoid arthritis. Rheumatology (Oxford) 2005, 44: 949–50.

    Article  CAS  Google Scholar 

  44. Ito K, Hori K, Terashima Y, Sekine M, Kura H. Insufficiency fracture of the body of the calcaneus in elderly patients with osteoporosis: a report of two cases. Clin Orthop Relat Res 2004, 422: 190–4.

    Article  PubMed  Google Scholar 

  45. Franco M, Albano L, Kacso I, Gaïd H, Jaeger P. An uncommon cause of footpain: the cuboid insufficiency stress fracture. Joint Bone Spine 2005, 72: 76–8.

    Article  PubMed  Google Scholar 

  46. Tomczak RL, VanCourt R. Metatarsal insufficiency fractures in previously undiagnosed osteoporosis patients. J Foot Ankle Surg 2000, 39: 174–83.

    Article  PubMed  CAS  Google Scholar 

  47. Kaye RA. Insufficiency stress fractures of the foot and ankle in postmenopausal women. Foot Ankle Int 1998, 19: 221–4.

    Article  PubMed  CAS  Google Scholar 

  48. Min JK, Sung MS. Insufficiency fractures of the sternum. Scand J Rheumatol 2003, 32: 179–80.

    Article  PubMed  CAS  Google Scholar 

  49. Benbouazza K, Harzy T, Guedira N, Hajja-Hassouni N. Insufficiency fracture of the sternum report of a case. Joint Bone Spine 2003, 70: 87–8.

    Article  PubMed  Google Scholar 

  50. Min JK, Joo HJ, Kim TH, et al. A case of sternal insufficiency fracture. Korean J Intern Med 1999, 14: 94–7.

    PubMed  CAS  Google Scholar 

  51. Maraval A, Grados F, Royant V, Damade R, Boulu G, Fardellone P. Longitudinal femoral shaft fracture due to bone insufficiency. A review of three cases. Joint Bone Spine 2003, 70: 526–31.

    Article  Google Scholar 

  52. Williams M, Laredo JD, Setbon S, et al. Unusual longitudinal stress fractures of the femoral diaphysis: report of five cases. Skelet Radiol 1999, 28: 81–5.

    Article  CAS  Google Scholar 

  53. Soubrier M, Dubost JJ, Boisgard S, et al. Insufficiency fracture. A survey of 60 cases and review of the literature. Joint Bone Spine 2003, 70: 209–18.

    Article  Google Scholar 

  54. Buskila D, Gladman DD. Stress fractures of the legs and swelling of the ankles in a patient with lupus: a diagnostic dilemma. Ann Rheum Dis 1990, 49: 783–4.

    Article  PubMed Central  PubMed  CAS  Google Scholar 

  55. Tarr RW, Kaye JJ, Nance EP Jr. Insufficiency fractures of the femoral neck in association with chronic renal failure. South Med J 1988, 81: 863–6.

    Article  PubMed  CAS  Google Scholar 

  56. Milgrom C, Finestone A, Shlamkovitch N, et al. Youth is a risk factor for stress fracture. A study of 783 infantry recruits. J Bone Joint Surg Br 1994, 76: 20–2.

    CAS  Google Scholar 

  57. Jung JH, Jun JB, Shim SC, et al. A case of parasymphyseal and associated insufficiency fractures of pubic rami in a patient with mixed connective tissue disease. Korean J Intern Med 2000, 15: 160–3.

    PubMed  CAS  Google Scholar 

  58. Elkayam O, Paran D, Flusser G, Wigler I, Yaron M, Caspi D. Insufficiency fractures in rheumatic patients: misdiagnosis and underlying characteristics. Clin Exp Rheumatol 2000, 18: 369–74.

    PubMed  CAS  Google Scholar 

  59. Manaster BJ, Dalinka MK, Alazraki N, et al. Stress/insufficiency fractures (excluding vertebral). American College of Radiology. ACR Appropriateness Criteria. Radiology 2000, 215: 265–72.

    Google Scholar 

  60. Blake SP, Connors AM. Sacral insufficiency fracture. Br J Radiol 2004, 77: 891–6.

    Article  PubMed  CAS  Google Scholar 

  61. White JH, Hague C, Nicolaou S, Gee R, Marchinkow LO, Munk PL. Imaging of sacral fractures. Clin Radiol 2003, 58: 914–21.

    Article  PubMed  CAS  Google Scholar 

  62. Boniotti V, Del Giudice E, Fengoni E, Cerini R, Caudana R. Imaging of bone micro-injuries. Radiol Med (Torino) 2003, 105: 425–35.

    Google Scholar 

  63. Soubrier M, Dubost JJ, Boisgard S, et al. Insufficiency fracture. A survey of 60 cases and review of the literature. Joint Bone Spine 2003, 70: 209–18.

    Article  Google Scholar 

  64. Chen CK, Liang HL, Lai PH, et al. Imaging diagnosis of insufficiency fracture of the sacrum. Zhonghua Yi Xue Za Zhi (Taipei) 1999, 62: 591–7.

    CAS  Google Scholar 

  65. Takahara K, Kamimura M, Nakagawa H, Uchiyama S. Changes in biochemical markers of bone in patients with insufficiency fractures. J Bone Miner Metab 2004, 22: 618–25.

    Article  PubMed  Google Scholar 

  66. Taillandier J, Langue F, Alemanni M, Taillandier-Heriche E. Mortality and functional outcomes of pelvic insufficiency fractures in older patients. Joint Bone Spine 2003, 70: 287–9.

    Article  PubMed  Google Scholar 

  67. Dasgupta B, Shah N, Brown H, Gordon TE, Tanqueray AB, Mellor JA. Sacral insufficiency fractures: an unsuspected cause of low back pain. Br J Rheumatol 1998, 37: 789–93.

    Article  PubMed  CAS  Google Scholar 

  68. Martin-Hunyadi C, Heitz D, Kaltenbach G, et al. Spontaneous insufficiency fractures of long bones: a prospective epidemiological survey in nursing home subjects. Arch Gerontol Geriatr 2000, 31: 207–14.

    Article  PubMed  Google Scholar 

  69. Martin-Hunyadi C, Kaltenbach G, Heitz D, Demuynck-Roegel C, Berthel M, Kuntzmann F. Clinical and prognostic aspects of spontaneous fractures in long term care units: a thirty month prospective study. Eastern Gerontology Society. Rev Med Interne 2000, 21: 747–55.

    Article  CAS  Google Scholar 

  70. Odvina CV, Zerwekh JE, Rao DS, Maalouf N, Gottschalk FA, Pak CY. Severely suppressed bone turnover: a potential complication of alendronate therapy. J Clin Endocrinol Metab 2005, 90: 1294–301.

    Article  PubMed  CAS  Google Scholar 

  71. Meurman KO, Elfving S. Stress fracture in soldiers: a multifocal bone disorder. A comparative radiological and scintigraphic study. Radiology 1980, 134: 485–7.

    Google Scholar 

  72. Matheson GO, Clement DB, McKenzie DC. Stress fractures in athletes: a study of 320 cases. Am J Sports Med 1987, 15: 46–58.

    Article  PubMed  CAS  Google Scholar 

  73. Schmid L, Pfirrmann C, Hess T, Schlumpf U. Bilateral fracture of the sacrum associated with pregnancy: a case report. Osteoporos Int 1999, 10: 91–3.

    Article  PubMed  CAS  Google Scholar 

  74. Breuil V, Brocq O, Euller-Ziegler L, Grimaud A. Insufficiency fracture of the sacrum revealing a pregnancy associated osteoporosis. First case report. Ann Rheum Dis 1997, 56: 278–9.

    Article  CAS  Google Scholar 

  75. Ubara Y, Higa Y, Tagami T, et al. Pelvic insufficiency fracture related to autosomal dominant polycystic kidney disease. Am J Kidney Dis 2005, 46: e103–11.

    Article  PubMed  Google Scholar 

  76. Zarrinkalam KH, Kuliwaba JS, Martin RB, Wallwork MA, Fazzalari NL. New insights into the propagation of fatigue damage in cortical bone using confocal microscopy and chelating fluorochromes. Eur J Morphol 2005, 42: 81–90.

    Article  PubMed  CAS  Google Scholar 

  77. Kruzic JJ, Scott JA, Nalla RK, Ritchie RO. Propagation of surface fatigue cracks in human cortical bone. J Biomech 2006, 39: 968–72.

    Article  PubMed  CAS  Google Scholar 

  78. Khosla S, Melton LJ 3rd, Achenbach SJ, Oberg AL, Riggs BL. Hormonal and biochemical determinants of trabecular microstructure at the ultradistal radius in women and men. J Clin Endocrinol Metab 2006, 91: 885–91.

    Article  PubMed  CAS  Google Scholar 

  79. Stampa B, Kuhn B, Liess C, Heller M, Gluer CC. Characterization of the integrity of three-dimensional trabecular bone microstructure by connectivity and shape analysis using high-resolution magnetic resonance imaging in vivo. Top Magn Reson Imaging 2002, 13: 357–63.

    Article  PubMed  Google Scholar 

  80. O’Brien FJ, Brennan O, Kennedy OD, Lee TC. Microcracks in cortical bone: how do they affect bone biology? Curr Osteoporos Rep 2005, 3: 39–45.

    Article  PubMed  Google Scholar 

  81. Cranney A, Guyatt G, Griffith L, Wells G, Tugwell P, Rosen C. Osteoporosis Methodology Group and The Osteoporosis Research Advisory Group. Meta-analyses of therapies for postmenopausal osteoporosis. IX: Summary of meta-analyses of therapies for postmenopausal osteoporosis. Endocr Rev 2002, 23: 570–8.

    CAS  Google Scholar 

  82. Zafran N, Liss Z, Peled R, Sherf M, Reuveni H. Incidence and causes for failure of treatment of women with proven osteoporosis. Osteoporos Int 2005, 16: 1375–83.

    Article  PubMed  Google Scholar 

  83. Lo JC, Pressman AR, Omar MA, Ettinger B. Persistence with weekly alendronate therapy among postmenopausal women. Osteoporos Int. 2006, 17: 922–8.

    Article  PubMed  CAS  Google Scholar 

  84. Rossini M, Bianchi G, Di Munno O, et al. Determinants of adherence to osteoporosis treatment in clinical practice. Osteoporos Int 2006, 17: 914–21.

    Article  PubMed  CAS  Google Scholar 

  85. Rosen HN, Moses AC, Garber J, Ross DS, Lee SL, Greenspan SL. Utility of biochemical markers of bone turnover in the fol low-up of patients treated with bisphosphonates. Calcif Tissue Int 1998, 63: 363–8.

    Article  PubMed  CAS  Google Scholar 

  86. Rosen HN, Moses AC, Garber J, et al. Serum CTX: a new marker of bone resorption that shows treatment effect more often than other markers because of low coefficient of variability and large changes with bisphosphonate therapy. Calcif Tissue Int 2000, 66: 100–3.

    Article  PubMed  CAS  Google Scholar 

  87. Ravn P, Hosking D, Thompson D, et al. Monitoring of alendronate treatment and prediction of effect on bone mass by biochemical markers in the early postmenopausal intervention cohort study. J Clin Endocrinol Metab 1999, 84: 2363–8.

    PubMed  CAS  Google Scholar 

Download references

Author information

Authors and Affiliations

Authors

Corresponding author

Correspondence to P. Lee MBBS (Hons) University of Melbourne.

Rights and permissions

Reprints and permissions

About this article

Cite this article

Lee, P., van der Wall, H. & Seibel, M.J. Looking beyond low bone mineral density: Multiple insufficiency fractures in a woman with post-menopausal osteoporosis on alendronate therapy. J Endocrinol Invest 30, 590–597 (2007). https://doi.org/10.1007/BF03346353

Download citation

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/BF03346353

Key-words

Navigation