Skip to main content
SpringerLink
Log in

Management of nerve compression in multiple hereditary exostoses: a report of two cases and review of the literature

  • Case Report
  • Published:
Child's Nervous System Aims and scope Submit manuscript

Abstract

Purpose

Multiple hereditary exostoses (MHE) is a rare autosomal dominant condition that results in the growth of cartilage-capped prominences that often cause nerve compression and injury. Many patients suffer from continued and debilitating chronic pain which leads some to advocate avoiding surgical intervention in patients with multiple hereditary exostoses. We present a review of the literature as well as a case series at our institution in order to evaluate the role of surgery in multiple hereditary exostoses.

Methods

We searched the literature for reports of patients with multiple hereditary exostoses undergoing surgery for nerve compression. We then reviewed the recent experience at our institution which revealed two patients with multiple hereditary exostoses.

Results

Our literature search revealed that there have been several case series and retrospective analyses in the literature that assess the benefit of surgery in the case of nerve compression caused by exostoses. The majority of these reports are of solitary exostoses. Few reports expand on the role of surgery in patients with multiple hereditary exostoses suffering from nerve compressions secondary to bony overgrowth. A recent review of the experience at our institution revealed two patients with multiple hereditary exostoses who together underwent a total of four surgeries for treatment of peripheral nerve compression resulting in pain or weakness. Postoperative evaluation revealed improvement in pain and/or motor strength following each operation.

Conclusion

Based on our experience and literature review, we advocate that nerve compression in selected individuals with multiple hereditary exostoses that results in neurological injury should be considered for nerve decompression and resection of the offending exostosis.

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. 1
Fig. 2

Similar content being viewed by others

References

  1. Pannier S, Legeai-Mallet L (2008) Hereditary multiple exostoses and enchondromatosis. Best Pract Res Clin Rheumatol 22:45–54. doi:10.1016/j.berh.2007.12.004

    Article  CAS  PubMed  Google Scholar 

  2. Stieber JR, Dormans JP (2005) Manifestations of hereditary multiple exostoses. J Am Acad Orthop Surg 13:110–120

    Article  PubMed  Google Scholar 

  3. Marshall HW (1916) A case of multiple cartilaginous exostoses. JBJS Case Connect 14:346–359

    Google Scholar 

  4. Legeai-Mallet L, Munnich A, Maroteaux P, Le Merrer M (1997) Incomplete penetrance and expressivity skewing in hereditary multiple exostoses. Clin Genet 52:12–16

    Article  CAS  PubMed  Google Scholar 

  5. Flores LP, Koerbel A, Tatagiba M (2005) Peroneal nerve compression resulting from fibular head osteophyte-like lesions. Surg Neurol 64:249–252. doi:10.1016/j.surneu.2004.11.031

    Article  PubMed  Google Scholar 

  6. Slepian A, Hamby WB (1951) Neurologic complications associated with hereditary deforming chondrodysplasia: review of the literature and a report of two cases occurring in the same family. J Neurosurg 8:529–535

    Article  CAS  PubMed  Google Scholar 

  7. Göçmen S, Topuz AK, Atabey C, et al. (2014) Peripheral nerve injuries due to osteochondromas: analysis of 20 cases and review of the literature. J Neurosurg 120:1105–1112. doi:10.3171/2013.11.JNS13310

    Article  PubMed  Google Scholar 

  8. Wicklund CL, Pauli RM, Johnston D, Hecht JT (1995) Natural history study of hereditary multiple exostoses. Am J Med Genet 55:43–46. doi:10.1002/ajmg.1320550113

    Article  CAS  PubMed  Google Scholar 

  9. Burki V, SO A, Aubry-Rozier B (2011) Cervical myelopathy in hereditary multiple exostoses. Jt Bone Spine 78:412–414. doi:10.1016/j.jbspin.2011.02.021

    Article  Google Scholar 

  10. Aldea S, Bonneville F, Poirier J, et al. (2006) Acute spinal cord compression in hereditary multiple exostoses. Acta Neurochir (Wien) 148:195–198. doi:10.1007/s00701-005-0680-6

    Article  CAS  Google Scholar 

  11. Bess RS, Robbin MR, Bohlman HH, Thompson GH (2005) Spinal exostoses: analysis of twelve cases and review of the literature. Spine (Phila Pa 1976) 30:774–780. doi:10.1097/01.brs.0000157476.16579.a2

    Article  Google Scholar 

  12. Gray KV, Robinson J, Bernstein RM, Otsuka NY (2004) Splitting of the common peroneal nerve by an osteochondroma: two case reports. J Pediatr Orthop Part B / Eur Paediatr Orthop Soc Pediatr Orthop Soc North Am 13:281–283. doi:10.1097/01.bpb.0000111022.13276.0a

    Google Scholar 

  13. Govender S, Parbhoo AH (1999) Osteochondroma with compression of the spinal cord. A report of two cases. J Bone Joint Surg Br 81:667–669. doi:10.1302/0301-620X.81B4.9248

    Article  CAS  PubMed  Google Scholar 

  14. Paik NJ, Han TR, Lim SJ (2000) Multiple peripheral nerve compressions related to malignantly transformed hereditary multiple exostoses. Muscle Nerve 23:1290–1294. doi:10.1002/1097-4598(200008)23:8<1290:AID-MUS23>3.0.CO;2-M

    Article  CAS  PubMed  Google Scholar 

  15. Nunez DA, Graham HK, Fixsen JA (1990) Cervical cord compression and femoral neuropathy in hereditary multiple exostoses. J Neurol Neurosurg Psychiatry 53:439. doi:10.1136/jnnp.53.5.439

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  16. Ashraf A, Larson AN, Ferski G, et al. (2013) Spinal stenosis frequent in children with multiple hereditary exostoses. J Child Orthop 7:183–194. doi:10.1007/s11832-013-0484-9

    Article  PubMed  PubMed Central  Google Scholar 

  17. Tian Y, Yuan W, Chen H, Shen X (2011) Spinal cord compression secondary to a thoracic vertebral osteochondroma. J Neurosurg Spine 15:252–257. doi:10.3171/2011.4.SPINE10484

    Article  PubMed  Google Scholar 

  18. Zaijun L, Xinhai Y, Zhipeng W, et al. (2013) Outcome and prognosis of myelopathy and radiculopathy from osteochondroma in the mobile spine: a report on 14 patients. J Spinal Disord Tech 26:194–199. doi:10.1097/BSD.0b013e31823eb239

    Article  PubMed  Google Scholar 

  19. Goud AL, de Lange J, Scholtes VAB, et al. (2012) Pain, physical and social functioning and quality of life in individuals with multiple hereditay exostoses in the Netherlands. J Bone Jt Surg 94:1013. doi:10.2106/JBJS.K.00406

    Article  CAS  Google Scholar 

  20. Darilek S, Wicklund C, Novy D, et al. (2005) Hereditary multiple exostosis and pain. J Pediatr Orthop 25:369–376. doi:10.1097/01.bpo.0000150813.18673.ad

    Article  PubMed  Google Scholar 

  21. Bottner F, Rodl R, Kordish I, et al. (2003) Surgical treatment of symptomatic osteochondroma. A three-to eight-year follow-up study. J Bone Joint Surg Br 85:1161–1165. doi:10.1302/0301-620X.85B8.14059

    Article  CAS  PubMed  Google Scholar 

  22. Cardelia JM, Dormans JP, Drummond DS, et al. (1995) Proximal fibular osteochondroma with associated peroneal nerve palsy: a review of six cases. J Pediatr Orthop 15:574–577. doi:10.1097/01241398-199509000-00004

    Article  CAS  PubMed  Google Scholar 

  23. Karakurt L, Yilmaz E, Varol T, et al. (2004) Solitary osteochondroma of the elbow causing ulnar nerve compression: a case report. Acta Orthop Traumatol Turc 38:291–294

    PubMed  Google Scholar 

  24. Yu K, Meehan JP, Fritz A, Jamali AA (2010) Osteochondroma of the femoral neck: a rare cause of sciatic nerve compression. Orthopedics. doi:10.3928/01477447-20100625-26

    Google Scholar 

  25. Murphy SM, Browne K, Tuite DJ, O’Shaughnessy M (2013) Dual pathology proximal median nerve compression of the forearm. J Plast Reconstr Aesthet Surg 66:1792–1794. doi:10.1016/j.bjps.2013.04.054

    Article  PubMed  Google Scholar 

  26. Turan Ilica A, Yasar E, Tuba Sanal H, et al. (2008) Sciatic nerve compression due to femoral neck osteochondroma: MDCT and MR findings. Clin Rheumatol 27:403–404. doi:10.1007/s10067-007-0761-4

    Article  PubMed  Google Scholar 

  27. Ryckx A, Somers JF, Allaert L (2013) Hereditary multiple exostosis. Acta Orthop Belg 79:597–607

    PubMed  Google Scholar 

  28. Scmale GA, Conrad EU, Raskind WH (1994) The natural history of hereditary multiple exostoses. J Bone Joint Surg Am 76:986–992

    Article  Google Scholar 

  29. Jones KB (2012) Glycobiology and the growth plate: current concepts in multiple hereditary exostoses. J Pediatr Orthop 31:577–586. doi:10.1097/BPO.0b013e31821c7738

    Article  Google Scholar 

  30. Muthusamy S, Conway SA, HT T (2014) Five polyostotic conditions that general orthopedic surgeons should recognize (or should not miss). Orthop Clin North Am 45:417–429. doi:10.1016/j.ocl.2014.04.004

    Article  PubMed  Google Scholar 

  31. Bovee JVMG (2008) Multiple osteochondromas. Orphanet J Rare Dis 3:3. doi:10.1186/1750-1172-3-3

    Article  PubMed  PubMed Central  Google Scholar 

  32. Czajka CM, DiCaprio MR (2015) What is the proportion of patients with multiple hereditary exostoses who undergo malignant degeneration? Clin Orthop Relat Res. doi:10.1007/s11999-015-4134-z

    PubMed  PubMed Central  Google Scholar 

  33. Çınar A, Yumrukçal F, Salduz A, et al. (2014) A rare cause of “drop foot” in the pediatric age group: proximal fibular osteochondroma a report of 5 cases. Int J Surg Case Rep 5:1068–1071. doi:10.1016/j.ijscr.2014.09.024

    Article  PubMed  PubMed Central  Google Scholar 

  34. Yoo JH, Min KD, Kim CK, Cha JG (2010) A case of extension loss of great toe due to peroneal nerve compression by an osteochondroma of the proximal fibula. Arch Orthop Trauma Surg 130:1071–1075. doi:10.1007/s00402-009-0944-3

    Article  PubMed  Google Scholar 

  35. Levin KH, Wilbourn AJ, Jones HR (1991) Childhood peroneal neuropathy from bone tumors. Pediatr Neurol 7:308–309. doi:10.1016/0887-8994(91)90053-N

    Article  CAS  PubMed  Google Scholar 

  36. Mnif H, Koubaa M, Zrig M, et al. (2009) Peroneal nerve palsy resulting from fibular head osteochondroma. Orthopedics 32:528. doi:10.3928/01477447-20090527-24

    Article  PubMed  Google Scholar 

  37. Levine PA, Frierson HF, Stewart FM, et al. (1987) Sinonasal undifferentiated carcinoma: a distinctive and highly aggressive neoplasm. Laryngoscope 97:905–908

    CAS  PubMed  Google Scholar 

Download references

Acknowledgments

No financial or material support has been received.

Author information

Authors and Affiliations

  1. Department of Neurosurgery, Penn State Milton S. Hershey Medical Center, 30 Hope Drive, Building B, Suite 1200, Hershey, PA, 17033, USA

    Russell Payne, Emily Sieg, Kimberly Harbaugh & Elias Rizk

  2. Department of Orthopedics, Pennsylvania State Milton S. Hershey Medical Center, Hershey, PA, USA

    Edward Fox

Authors
  1. Russell Payne
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Emily Sieg
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Edward Fox
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. Kimberly Harbaugh
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. Elias Rizk
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to Russell Payne.

Ethics declarations

Conflict of interest

The authors have nothing to disclose.

Additional information

Portions of this work were present as a poster at the 2015 Pennsylvania Neurosurgical Society meeting in Hershey, PA.

Rights and permissions

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Payne, R., Sieg, E., Fox, E. et al. Management of nerve compression in multiple hereditary exostoses: a report of two cases and review of the literature. Childs Nerv Syst 32, 2453–2458 (2016). https://doi.org/10.1007/s00381-016-3166-3

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00381-016-3166-3

Keywords

Search

Navigation