Azcarate, J. (J.)

Cañadell, J.M. (J. M.)

Imizcoz, A. (A.)

Martinez-de-Negri, J. (J.)

Villas-Tome, C. (Carlos)

Universidad de Navarra

Vertebral Eosinophilic Granuloma Long-Term Evolution of a Case

Dadun, University of Navarra

    Vertebral Eosinophilic Granuloma Long-Term Evolution of a Case   J. Cañadell, C. Villas, J. Martínez-Denegri, J. Azcarate, and A. Imizcoz   Address reprint requests to Professor J. Cañadell  Department of Orthopaedic Surgery and Traumatology, University Clinic Faculty of Medicine, University of Navarra Pamplona, Spain         INTRODUCTION  The term “vertebra plana” corresponds to the condition described by Calvé4 in 1925 as vertebral osteochondritis. In 1940 Otani and Ehrlich17 named it solitary granuloma of the bone. Later, other authors5,8,12 demonstrated that the so-called vertebra plana did not correspond to a vertebral osteochondritis but to eosinophilic granuloma affecting the spine. Other authors upheld such hypotheses.3,14,25  Schajowicz22 reported that the incidence of vertebral eosinophilic granuloma represents 7.8% of the total solitary eosinophilic granuloma and the Netherlands Committee on Bone Tumors16 reported an incidence of 15%. In spite of the varying statistical reports, the vertebral localization of this tumor cannot be considered infrequent.  Although neurologic involvement has been reported,9 clinical symptoms generally include pain and spasm of the paravertebral muscles, especially in children.4  Curettage of the lesion with or without bone grafting has been recommended as the management of choice.7,16,20,23 Nevertheless, a conservative approach is considered sufficient in treating vertebral eosinophilic granuloma11,13,19,24 except for those cases with neurologic involvement. There is no general agreement with respect to the efficacy of radiotherapy.19,22  The purpose of this study is to present a case of eosinophilic granuloma that was managed conservatively and that was followed closely for 15 years.   CASE REPORT  A white boy, born January 1965, came to our clinic in May 1969. The parents had noticed that he had difficulty moving about. During the last 45 days the patient complained of pain in the left hip. There was no relevant personal family history. No significant findings were appreciated upon inspection. Physical examination showed right genual hyperreflexia, lumbar hyperlordosis, difficulty in sitting up, and evidence of Gower's sign.   Radiographs of the spine showed collapse of L1 with loss of 75% of the vertebral body height (Figure 1). A needle biopsy confirmed the suspicion of vertebral eosinophilic granuloma.  The patient was treated initially with a plaster cast. Later, a Milwaukee brace was used for 3 months. Against advice, the patient discontinued the treatment. Evolution was followed for 4 years; the patient did not consult us again until 1984, when he presented with a slight retraction of the hamstrings. The patient did not complain of pain or weakness.  Radiographs of the spine showed reconstitution of the L1 vertebral body height with slight platyspondylia (vertebral body height, 92.59%). Figure 2 shows the first, fourth and fifteenth year of the disease's evolution.   DISCUSSION  Although eosinophilic granuloma is the commonest cause of vertebra plana,5,6,12-14,16,22-24 other pathologic conditions must be kept in mind because of their appearance in radiologic studies.10,18,21 In our opinion, biopsy is mandatory for an accurate diagnosis. Clinical evolution, the radiographic picture, and histopathologic signs coincided in our patient.  As refered to by Ippolito11 et al only a few patients with vertebra plana have been followed to skeletal maturity. Globally, there are no significant differences among the published cases1,11,15,38 and the one we present here.  At the T12-L1 level, the vertebral body flattening due to eosinophilic granuloma can theoretically induce angular kyphosis; nevertheless, our case and those reported by Ippolito11 et al have followed a satisfactory evolution, even when we consider that two patients discontinued using the Milwaukee brace.  Although the remodeling process of vertebra plana is not well known, our case and the reported experiences seem to support that the results are good whatever the treatment.7 Thus, we think surgery is indicated only in those cases with neurologic involvement.9  REFERENCES  1. Ankerhold J: Beobachtungen zum Verlauf des Calvéschen Plattwirbels. Arch Orthop Unfall-Chir 64:101-110, 1968 2. Buxton StJD: Spinal disease complicating Gaucher's disease in a child. Proc R Soc Med 28:1027-1028, 1935 3. Calhoun JD, Thompson SB: Vertebra plana in children produced by xanthomatous disease. AJR 82:482-489, 1959 4. Calvé JA: Localized affection of the spine suggesting osteocondritis of the vertebral body with clinical aspects of Pott's disease. J Bone Joint Surg 7:41-46, 1925 5. Compere EL, Johnson WE, Coventry MB: Vertebra plana (Calvé's disease) due to eosinophilic granuloma. J Bone Joint Surg 36A:969-980, 1954 6. Epstein BS: Afecciones de la Columna Vertebral y de la Médula Espinal. Barcelona, Ed Jims, 1973 7. Fowles JN, Bobechko NP: Solitary eosinophilic granuloma in bone. J Bone Joint Surg 52B:238-243, 1970 8. Green NT, Farber D: Eosinophilic or solitary granuloma of bone. J Bone Joint Surg 24:499-525, 1942 9. Green NE, Robertson WW, Kilroy AW: Eosinophilic granuloma of the spine with associated neural deficit: Report of three cases. J Bone Joint Surg 62A:1198-1202, 1980 10. Hillman JW: Discussion of Compere et al. J Bone Joint Surg 36A:978-979, 19545 11. Ippolito E, Farsetti P, Tudisco C: Vertebra plana. Long-term follow-up in five patients. J Bone Joint Surg 66A:1364-1368, 1984 12. Jaffe HL, Lichtenstein L: Eosinophilic granuloma of bone. Arch Pathol 37:99-118, 1944 13. McCullough CJ: Eosinophilic granuloma of bone. Acta Orthop Scand 51:389-398, 1980 14. Moseley JE: Patterns of bone changes in the reticuloendothelioses. J Mt Sinai Hosp 29:282-321, 1963 15. Nesbit NE, Kieffer SD, A'Angio GJ: Reconstitution of vertebral height in histiocytosis X: A long-term follow-up. J Bone Joint Surg 51A:1360-1368, 1969 16. Netherland Committee on Bone Tumors: Radiological Atlas of Bone Tumors, Vol II. The Hague, Mouton and Co, 1973, pp 501-504 17. Otani S, Ehrlich J: Solitary granuloma of bone simulating primari neoplasm. Am J Pathol 16:479-490, 1940 18. Poulsen JO, Jensen JT, Thommesen P: Ewing's sarcoma simulating vertebra plana. Acta Orthop Scand 46:211-215, 1975 19. Ruff SJ, Taylor TK, Nicholson OR: Eosinophilic granuloma of the spine. J Bone Joint Surg 66B:780, 1984 20. Sbarbaro JC, Kenneth CF: Eosinophilic granuloma of bone. JAMA 178:706-710, 1961 21. Schachar NS, Edwards GE: Vertebra plana (Calvé's disease) due to aneurysmal bone cyst. J Bone Joint Surg 56B:586, 1974 22. Schajowicz F: Tumores y Lesiones Pseudotumorales de Huesos y Articulaciones. Buenos Aires, Ed Panamericana, 1982, pp 464-481 23. Tachdjian MO: Ortopedia Pediátrica. Vol I. México, Interamericana, 1972, p 54 24. Turek S: Ortopedia, Principios y Aplicaciones. Vol I. Barcelona, Ed Salvat, 1982, p 793 25. Virenque J, Pasquié M, Glaubert J: Notes sur le ganulome des os. Presse Med 68:2304-2037, 1960        Figure 1. Collapse of L1 with loss of 75% of vertebral body height in 4-year old boy.        Figure 2. Vertebral eosinophilic granuloma progress at year 1, 4, and 15, respectively.     

Vertebral Eosinophilic Granuloma Long-Term Evolution of a Case

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