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Eosinophilic Granuloma Of  The Ilium -A Case Report

 M. Shantharam Shetty*, Ajith Kumar**, Sandeep S. Ireshanavar***

* HOD Department of Orthopaedics, Tejasvini Hospital
** Consultant Orthopaedic Surgeon, Tejasvini Hospital
*** Registrar In Orthopaedics, Tejasvini Hospital

Address for Correspondence:
Prof. M.  Shantharam Shetty
Tejasvini Hospital And SSIOT
Kadri,  Mangalore , Karnataka,India
Phone: 0824 2225777, 2225996, 2225997
Fax: 0824 2225998


Localised Langerhans-cell histiocytosis of bone (eosinophilic granuloma) is a benign tumour-like condition with a variable clinical course. Different forms of treatment have been reported to give satisfactory results. Although the mechanism of action of intralesional injection of a corticosteroid has not been defined, use of percutaneous needle biopsy to diagnose localized Langerhans-cell histiocytosis and treatment with intralesional administration of methylprednisolone relieved pain expeditiously, enabled the patient to avoid an operative procedure, and resulted in osseous healing. Right Ilium was involved in a 3 year old boy. CT guided biopsy revealed eosinophilic granuloma.  Intralesional steroid instillation was done, lesion healed well with no recurrence after 7 years follow up.
Keywords: Eosinophilic Granuloma, Pelvis, Intralesional steroid

J.Orthopaedics 2007;4(2)e25



The clinical spectrum of Langerhans-cell histiocytosis (LCH) is wide, ranging from a potentially lethal leukaemia-like disorder to a solitary lytic lesion of bone10. Localised LCH of bone is a benign tumour-like condition which is characterised by a clonal proliferation of Langerhans-type histiocytes20, and is commonly referred to as eosinophilic granuloma12. The bones which are the most commonly involved are the skull, the pelvis, and the diaphysis of long bones18. The systemic condition includes Hand-Schüller-Christian disease with a triad of exophthalmos, diabetes insipidus and osteolytic lesions of the skull, and Letterer-Siwe disease with hepatosplenomegaly, lymphadenopathy, skin rash, fever, anaemia and thrombocytopenia12.

Case Report:

3year old boy presented to us with pain & swelling in right iliac region of 2 months duration. History of irregular fever and unable to bear weight on affected limb. The child was on antibiotic treatment before coming to our centre - being treated as osteomyelitis.


  • Right Hip in 30° fixed flexion deformity.

  • Severe tenderness over Right Iliac crest and gluteal region.

  • Severe painful restriction of (Rt) hip movements.

  • No distal Neurovascular Deficit.

  • General and systemic examination was normal. [FIGURE 1]

X-RAY: Pelvis revealed large Lytic lesion in Right Iliac bone [FIGURE 2] Serum Alkaline Phosphatase: 200 IU/L, rest all laboratory investigations were within normal limits.

CT scan reveled osteolytic lesion of the right ilium [FIGURE 3 AND 4]. At the same sitting CT guided FNAC was done with a wide bore needle. FNAC revealed Sheets of langerhan's cells. These cells are derived from the mononuclear cell and dendritic line precursors Cytoplasmic inclusion bodies --birbeck's granules suggestive of eosinophilic granuloma [FIGURE 5].

Patient underwent a single dose of intralesional 120 mg of methyl prednisolone acetate instillation. Pain free and improved movement at hip with  ability to bear weight  with in 3 weeks .Patient was followed periodically and there was total obliteration of the lesion at the end of 7 year follow up [FIGURES 6 AND 7] with no recurrence. 

Fig 1 Fig 2
Fig 3 Fig 4
Fig 5 Fig 6
Fig 7

Discussion :

Localized Langerhans-cell histiocytosis of bone (eosinophilic granuloma of bone) is a rare, benign tumorlike condition that is characterized by a clonal proliferation of Langerhans-type histiocytes 11,12,13,19. Although the etiology and pathogenesis remain unknown, the clinical course is usually benign but can be highly variable, with partial or complete healing of the lesion, recurrence after treatment, or progression or spontaneous remission without treatment 3,4,5.

Radiographically, the differential diagnosis includes osteomyelitis and Ewing sarcoma in children and metastatic carcinoma and other benign and malignant primary tumors of bone in adults. The diagnosis must be confirmed with operative (open) or percutaneous biopsy.

Osseous healing has been observed after various therapeutic interventions, including curettage or low-dose radiation therapy, or both5,14,16,17; intralesional injection of corticosteroids2,7,8,9; and oral chemotherapy 21.

Intralesional injection of corticosteroids is a non-operative approach that has been demonstrated to produce rapid relief of pain with predictable healing and without the need for additional intervention 2,7,8,9.

Although the precise mechanism of action of intralesional injection of methylprednisolone for the treatment of localized Langerhans-cell histiocytosis has not been defined, to our knowledge, several in vitro studies have demonstrated the production of interleukins (such as IL-1 [interleukin-1]) and prostaglandins (such as prostaglandins E2 and D2) by suspensions of Langerhans cells 1,6. The inhibition of IL-1-induced bone resorption and prostaglandin production by methylprednisolone may account for the dramatic response demonstrated both clinically and radiographically15. 


It is extremely difficult to establish guidelines for the treatment of a rare condition such as Langerhans-cell histiocytosis. The least invasive procedure appropriate for the level of expertise at a given institution. Intralesional steroid instillation when used gives rapid resolution of symptoms and in radiographic evidence of healing of the osseous lesions without recurrence.

Reference :

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  2. 2. Capanna R, Springfield DS, Ruggieri P, et al. Direct cortisone injection in eosinophilic granuloma of bone: a preliminary report on 11 patients. J Pediat Orthop 1985;5:339-42.

  3. 3. Chacha, P. B., and Khong, B. T.: Eosinophilic granuloma of bone. A diagnostic problem. Clin. Orthop., 80: 79-88, 1971.

  4. 4. den Herder, B. A.: Changing views on eosinophilic granuloma of bone. Radiol. Clin. Biol., 42: 218-221, 1973.

  5. 5. Fowles, J. V., and Bobechko, W. P.: Solitary eosinophilic granuloma in bone. J. Bone and Joint Surg., 52-B(2): 238-243, 1970

  6. 6. Gonzalez-Crussi, F.; Hsueh, W.; and Wiederhold, M. D.: Prostaglandins in histiocytosis-X. PG synthesis by histiocytosis-X cells. Am. J. Clin. Pathol., 75: 243-253, 1981.

  7. 7. Jones, L. R.; Toth, B. B.; and Cangir, A.: Treatment for solitary eosinophilic granuloma of the mandible by steroid injection: report of a case. J. Oral and Maxillofac. Surg., 47: 306-309, 1989.

  8. 8. Katz, R. L.; Silva, E. G.; DeSantos, L A.; and Lukeman, J. M.: Diagnosis of eosinophilic granuloma of bone by cytology, histology, and electron microscopy of transcutaneous bone-aspiration biopsy. J. Bone and Joint Surg., 62-A: 1284-1290, Dec. 1980.

  9. 9. Kindy-Degnan, N. A.; Laflamme, P.; Duprat, G.; and Allaire, G. S.: Intralesional steroid in the treatment of an orbital eosinophilic granuloma [letter]. Arch. Ophthalmol., 109: 617-618, 1991.

  10. 10. Ladish S, Jaffe ES. The histiocytocis. In: Pizzo PA, Poplack DG, eds. Principles and practices of pediatric oncology. Philadelphia: J. B. Lippincott Company, 1989:491-504

  11. 11. Lichtenstein, L.: Histiocytosis X: integration of eosinophilic granuloma of bone, "Letterer-Siwe" and "Schuller-Christian disease" as related manifestations of a single nosologic entity. Arch. Pathol., 56: 84-102, 1953.

  12. 12. Lichtenstein L. Histiocytosis X (eosinophilic granuloma of bone, 'Letterer-Siwe disease and Schuller-Christian disease'): further observations of pathological and clinical importance. J Bone Joint Surg [Am] 1964;46-A:76-90

  13. 13. Lichtenstein, L., and Jaffe, H. L.: Eosinophilic granuloma of bone. With report of a case. Am. J. Pathol., 16: 595-604, 1940.

  14. 14. Makley, J. T., and Carter, J. R.: Eosinophilic granuloma of bone. Clin. Orthop., 204: 37-44, 1986.

  15. 15. Marusc, A., and Raisz, L. G.: Cortisol modulates the actions of interleukin-1 alpha on bone formation, resorption, and prostaglandin production in cultured mouse parietal bones. Endocrinology, 129: 2699-2706, 1991.

  16. 16. McCullough, C. J.: Eosinophilic granuloma of bone. Acta Orthop. Scandinavica, 51: 389-398, 1980.

  17. 17. McGavran, M. H., and Spady, H. A.: Eosinophilic granuloma of bone. A study of twenty-eight cases. J. Bone and Joint Surg., 42-A: 979-992, Sept. 1960.

  18. 18. Nesbit ME Jr. Bone tumours in infants and children. Paediatrician 1972/73;1:273-87.

  19. 19. Schajowicz, F., and Slullitel, J.: Eosinophilic granuloma of bone and its relationship to Hand-Schuller-Christian and Letterer-Siwe syndromes. J. Bone and Joint Surg., 55-B(3): 545-565, 1973.

  20. 20. Willman CL, Busque L, Griffith BB, et al. Langerhans histiocytosis (histiocytosis X): a clonal proliferative disease. New Eng J Med 1994;331:154-60

  21. 21. Womer, R. B.; Anunciato, K. R.; and Chehrenama, M.: Oral methotrexate and alternate-day prednisone for low-risk Langerhans cell histiocytosis. Med. and Pediat. Oncol., 25: 70-73, 1995.


This is a peer reviewed paper 

Please cite as :M. Shantharam Shetty : Eosinophilic Granuloma Of  The Ilium -A Case Report

J.Orthopaedics 2007;4(2)e25





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