Skip to main content

Advertisement

SpringerLink
Log in

Eosinophilic granuloma of spine in adults: a report of 30 cases and outcome

  • Clinical Article
  • Published:
Acta Neurochirurgica Aims and scope Submit manuscript

Abstract

Background

Eosinophilic granuloma (EG) of the spine is rare, especially in adults. There had been few large and long-term studies reported in the literature. The management goals of this disease in adults are preservation of neurologic function, relief of pain and reconstruction of spinal stability. However, there are still controversies over appropriate management modality of eosinophilic granuloma.

Methods

Clinical manifestations, radiographic presentations, therapeutic outcomes and follow-up findings of 30 adults who were histiologically diagnosed with spinal eosinophilic granuloma, including 28 patients who received surgical treatment at our institutions from 1985 to 2008 were reviewed retrospectively.

Results

There were 25 males and five females with a mean age of 34.5 years (range, 18–71 years). The post-operative follow-up period ranged from 2 to 22.4 years (mean, 8.3 years). Neurologic deficits developed in 21 patients, apparent kyphosis developed in four cases. In contrast to the classic feature of vertebra plana in children, we found that more severe lesions often led to asymmetric collapse in adult patients and only three patients presented with vertebra plana. Thirty-three vertebral lesions distributed throughout the spine column. Twenty-one lesions were in cervical spine, seven in the thoracic spine and five in the lumbar spine. Twenty-eight adult patients underwent surgical resection with or without chemotherapy or radiotherapy, and four (13.3%) patients had recurrence after surgery. No patient in our series died.

Conclusions

The onset of spinal EG is insidious and mainly presents as osteolytic destruction. There is a particular high prevalence of lesions in the cervical spine and more severe lesions often led to asymmetric collapse. As the skeleton of adults is well-developed and the epiphysis has stopped growing, individualized management including surgical intervention should be considered in adult patients with spinal EG who present with neurological damage and spinal instability.

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
Fig. 3

Similar content being viewed by others

References

  1. Azouz EM, Saigal G, Rodriguez MM, Podda A (2005) Langerhans’ cell histiocytosis: pathology, imaging and treatment of skeletal involvement. Pediatr Radiol 35:103–115

    Article  PubMed  Google Scholar 

  2. Bertram C, Madert J, Eggers C (2002) Eosinophilic granuloma of the cervical spine. Spine 27:1408–1413

    Article  PubMed  Google Scholar 

  3. Binkovitz LA, Olshefski RS, Adler BH (2003) Coincidence FDG-PET in the evaluation of Langerhans’ cell histiocytosis: preliminary findings. Pediatr Radiol 33:598–602

    Article  PubMed  Google Scholar 

  4. Brown CW, Jarvis JG, Letts M, Carpenter B (2005) Treatment and outcome of vertebral Langerhans cell histiocytosis at the Children’s Hospital of Eastern Ontario. Can J Surg 48:230–236

    PubMed  Google Scholar 

  5. Capanna R, Springfield DS, Ruggieri P, Biaqini R, Picci P, Bacci G, Giunti A, Lorenzi EG, Campanacci M (1985) Direct cortisone injection in eosinophilic granuloma of bone. A preliminary report on 11 patients. J Pediatr Orthop 5:339–342

    CAS  PubMed  Google Scholar 

  6. Chu T, D’Angio GJ, Favara B, Ladisch S, Nesbit M, Pritchard J (1987) The writing group of the histiocyte society. Histiocytic syndromes in children. Lancet 1:208–209

    Google Scholar 

  7. Cohen M, Zornoza J, Cangir A, Murray JA, Wallace S (1980) Direct injection of methylprednisolone sodium succinate in the treatment of solitary eosinophilic granuloma of bone. A report of 9 cases. Radiology 136:289–293

    CAS  PubMed  Google Scholar 

  8. Denaro L, Longo UG, Papalia R, Di Martino A, Maffulli N, Denaro V (2008) Eosinophilic granuloma of the pediatric cervical spine. Spine 33:E936–941

    Article  PubMed  Google Scholar 

  9. Egeler RM, D’Angio GJ (1995) Langerhans cell histiocytosis. J Pediatr 127:1–11

    Article  CAS  PubMed  Google Scholar 

  10. Egeler RM, Thompson RC Jr, Voute PA, Nesbit ME Jr (1992) Intralesional infiltration of corticosteroids in localized Langerhans’ cell histiocytosis. J Pediatr Orthop 12:811–814

    CAS  PubMed  Google Scholar 

  11. Favara BE, Feller AC, Pauli M, Jaffe ES, Weiss LM, Arico M, Bucsky P, Egeler RM, Elinder G, Gadner H, Henter JI, Imashuku S, Janka-schaub G, Jaffe R, Ladisch S, Nezelof C, Pritchard J (1997) Contemporary classification of histiocytic disorders. The WHO Committee on Histiocytic/Reticulum Cell Proliferations. Reclassification Working Group of the Histiocyte Society. Med Pediatr Oncol 29:157–166

    Article  CAS  PubMed  Google Scholar 

  12. Fenoy AJ, Greenlee JD, Menezes AH, Donovan KA, Sato Y, Hitchon PW, Chaloupka JC (2006) Primary bone tumors of the spine in children. J Neurosurg 105(suppl 4):252–260

    PubMed  Google Scholar 

  13. Floman Y, Bar-On E, Mosheiff R, Mirovsky Y, Robin GC, Ramu N (1997) Eosinophilic granuloma of the spine. J Pediatr Orthop B 6:260–265

    CAS  PubMed  Google Scholar 

  14. Garg B, Sharma V, Eachempati KK, Malhotra R, Bhan S (2006) An unusual presentation of eosinophilic granuloma in an adult: a case report. J Orthop Surg 14:81–83

    CAS  Google Scholar 

  15. Garg S, Mehta S, Dormans JP (2004) Langerhans cell histiocytosis of the spine in children. Long-term follow-up. J Bone Joint Surg Am 86A:1740–1750

    Google Scholar 

  16. Garg S, Mehta S, Dormans JP (2003) An atypical presentation of Langerhans cell histiocytosis of the cervical spine in a child. Spine 28:E445–E448

    Article  PubMed  Google Scholar 

  17. Ghanem I, Tolo VT, D’Ambra P, Malogalowkin MH (2003) Langerhans cell histiocytosis of bone in children and adolescents. J Pediatr Orthop 23:124–130

    Article  PubMed  Google Scholar 

  18. Greenlee JD, Fenoy AJ, Donovan KA, Menezes AH (2007) Eosinophilic granuloma in the pediatric spine. Pediatr Neurosurg 43:285–292

    Article  PubMed  Google Scholar 

  19. Green WT, Farber S, McDermott LJ (1942) Eosinophilic or solitary Granuloma of bone. J Bone Jt Surg 24:499–526

    Google Scholar 

  20. Hindman BW, Thomas RD, Young LW, Yu L (1998) Langerhans cell histiocytosis: unusual skeletal manifestations observed in thirty four cases. Skeletal Radiol 27:177–181

    Article  CAS  PubMed  Google Scholar 

  21. Nakamura H, Nagayama R (2008) Eosinophilic granuloma present with local osteolysis in an adult lumbar spine. J Clin Nureosci 15:1398–1400

    Article  Google Scholar 

  22. Howarth DM, Gilchrist GS, Mullan BP, Wiseman GA, Edmonson JH, Schomberg PJ (1999) Langerhans cell histiocytosis: diagnosis, natural history, management, and outcome. Cancer 85:2278–2290

    Article  CAS  PubMed  Google Scholar 

  23. Ippolito E, Farsetti P, Tudisco C (1984) Vertebra plana. Long-term follow-up in five patients. J Bone Jt Surg Am 66:1364–1368

    CAS  Google Scholar 

  24. Islinger RB, Kuklo TR, Owens BD, Horan PJ, Choma TJ, Murphey MD, Temple HT (2000) Langerhans’ cell histiocytosis in patients older than 21 years. Clin Orthop Relat Res 379:231–235

    Article  PubMed  Google Scholar 

  25. Kaste SC, Rodriguex-Galindo C, McCarville ME, Shulkin BL (2007) PET-CT in pediatric Langerhans’s cell histiocytosis. Pediatr Radiol 37:615–622

    Article  PubMed  Google Scholar 

  26. Kilpatrick SE, Wenger DE, Gilchrist GS, Shives TC, Wollan PC, Unni KK (1995) Langerhans’ cell histiocytosis (histiocytosis X) of bone. A clinicopathologic analysis of 263 pediatric and adult cases. Cancer 76:2471–2484

    Article  CAS  PubMed  Google Scholar 

  27. Lauffenburger MD, Dull ST, Toselli R (1995) Eosinophilic granuloma of the adult spine: a case report and review of the literature. J Spinal Disord 8:243–248

    Article  CAS  PubMed  Google Scholar 

  28. Leiberman PH, Jones CR, Steinman RM, Erlandson RA, Smith J, Gee T, Huvos A, Garin-Chesa P, Filippa DA, Urmacher C, Ganqi MD, Sperber M (1996) Langerhans cell (eosinophilic) granulomatosis. A clinicopathologic study encompassing 50 years. Am J Surg Pathol 20:519–552

    Article  Google Scholar 

  29. Lichtenstein L (1953) Histiocytosis X. Integration of eosinophilicgranuloma of bone, “Letterer-Siwe disease”, and “Schiiller-Christian disease” as related manifestations of a single nosologic entity. Arch Pathol 56:84–102

    CAS  Google Scholar 

  30. Lichtenstein L, Jaffe ML (1940) Eosinophilic granuloma of bone, with a report of a case. Am J Pathol 16:595–604

    PubMed  CAS  Google Scholar 

  31. Malpas JS, Norton AJ (1996) Langerhans cell histiocytosis in the adult. Med Pediatr Oncol 27:540–546

    Article  CAS  PubMed  Google Scholar 

  32. Mammano S, Candiotto S, Balsano M (1997) Cast and brace treatment of eosinophilic granuloma of the spine: long-term follow-up. J Pediatr Orthop 17:821–827

    Article  CAS  PubMed  Google Scholar 

  33. Martinez-Lage JF, Poza M, Cartagena J, Vicente JP, Biec F, De Las HM (1991) Solitary eosinophilic granuloma of the pediatric skull and spine. The role of surgery. Child Nerv Syst 7:448–451

    Article  CAS  Google Scholar 

  34. Otani S, Ehrlich JC (1940) Solitary granuloma of bone simulating primary neoplasm. Am J Pathol 16:479–490

    PubMed  CAS  Google Scholar 

  35. Peng XS, Pan T, Chen LY, Huang G, Wang J (2009) Langerhans’ cell histiocytosis of the spine in children with soft tissue extension and chemotherapy. Int Orthop 33:731–736

    Article  PubMed  Google Scholar 

  36. Plasschaert F, Craig C, Bell R, Cole WG, Wunder JS, Alman BA (2002) Eosinophilic granuloma. A Different behaviour in children than in adults. J Bone Jt Surg Br 84-B:870–872

    Article  Google Scholar 

  37. Poulsen JO, Jensen JT, Nixon JE (1975) Ewing’s sarcoma simulating vertebra plana. Acta Orthop Scand 46:211–215

    Article  CAS  PubMed  Google Scholar 

  38. Raab P, Hohmann F, Kuhl J, Krauspe R (1998) Vertebral remodeling in eosinophilic granuloma of the spine. A long-term follow-up. Spine 23:1351–1354

    Article  CAS  PubMed  Google Scholar 

  39. Richter MP, D’Angio GJ (1981) The role of radiation therapy in the management of children with histiocytosis X. Am J Pediatr Hematol Oncol 3:161–163

    Article  CAS  PubMed  Google Scholar 

  40. Selch MT, Parker RG (1990) Radiation therapy in the management of Langerhans cell histiocytosis. Med Pediatr Oncol 18:97–102

    Article  CAS  PubMed  Google Scholar 

  41. Sessa S, Sommelet D, Lascombes P, Prevot J (1994) Treatment of Langerhans-cell histiocytosis in children: experience at the Children’s Hospital of Nancy. J Bone Jt Surg Am 76:1513–1525

    CAS  Google Scholar 

  42. Slater JM, Swarm OJ (1980) Eosinophilic granuloma of bone. Med Pediatr Oncol 8:151–164

    Article  CAS  PubMed  Google Scholar 

  43. Stull MA, Kransdorf MJ, Devaney KO (1992) Langerhans cell histiocytosis of bone. Radiographics 12:801–823

    CAS  PubMed  Google Scholar 

  44. Tan G, Samson I, De Wever I, Goffin J, Demaerel P, Van Gool SW (2004) Langerhans cell histiocytosis of the cervical spine: a single institution experience in four patients. J Pediatr Orthop 13:123–126

    Article  Google Scholar 

  45. Willis B, Ablin A, Weinberg V, Zoger S, Wara WM, Matthay KK (1996) Disease course and late sequelae of Langerhans’ cell histiocytosis: 25 year experience at the University of California, San Francisco. J Clin Oncol 14:2073–2082

    CAS  PubMed  Google Scholar 

  46. Yasko AW, Fanning CV, Ayala AG, Carrasco CH, Murray JA (1998) Percutaneous techniques for the diagnosis and treatment of localized Lanerhans-cell histiocytosis (eosinophilic granuloma of bone). J Bone Jt Surg Am 80:219–228

    CAS  Google Scholar 

  47. Yeom JS, Lee CK, Shin HY, Lee CS, Chang H (1999) Langerhans’ cell histiocytosis of the spine. Analysis of twenty-three cases. Spine 24:1740–1749

    Article  CAS  PubMed  Google Scholar 

Download references

Author information

Authors and Affiliations

  1. The Spine Center, Changzheng Hospital, Second Military Medical University, 415 Fengyang Road, Shanghai, 200003, China

    Wending Huang, Xinghai Yang, Dong Cao, Jianru Xiao, Mosong Yang, Dapeng Feng, Quan Huang, Zhipeng Wu, Wei Zheng & Lianshun Jia

  2. Department of Orthopaedic Surgery, Nanjing General Hospital of Nanjing Military Command, PLA, Nanjing, 210006, China

    Shujia Wu

Authors
  1. Wending Huang
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Xinghai Yang
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Dong Cao
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. Jianru Xiao
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. Mosong Yang
    View author publications

    You can also search for this author in PubMed Google Scholar

  6. Dapeng Feng
    View author publications

    You can also search for this author in PubMed Google Scholar

  7. Quan Huang
    View author publications

    You can also search for this author in PubMed Google Scholar

  8. Zhipeng Wu
    View author publications

    You can also search for this author in PubMed Google Scholar

  9. Wei Zheng
    View author publications

    You can also search for this author in PubMed Google Scholar

  10. Lianshun Jia
    View author publications

    You can also search for this author in PubMed Google Scholar

  11. Shujia Wu
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to Jianru Xiao.

Rights and permissions

Reprints and permissions

About this article

Cite this article

Huang, W., Yang, X., Cao, D. et al. Eosinophilic granuloma of spine in adults: a report of 30 cases and outcome. Acta Neurochir 152, 1129–1137 (2010). https://doi.org/10.1007/s00701-010-0644-3

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00701-010-0644-3

Keywords

Search

Navigation