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Print version ISSN 1130-1473

Neurocirugía vol.20 n.2  Apr. 2009




Orbital Langerhans cell histiocytosis with focus on treatment



Dear Editor:

We read with great interest the article by Ulivieri et al regarding the treatment of an orbital eosinophilic granuloma (EG). The authors treated a 25-year-old male harbouring an orbital EG by curettage of the lesion after bicoronal skin incision. Follow-up examinations revealed no evidence of recurrence5.

EG is the mildest form of Langerhans cell histiocytosis and usually affects children and young adults. The most common presentation is that of a solitary skeletal lesion, usually of the calvarium2. Surgical excision is the treatment mainstay. Nevertheless, other non-surgical treatments, such as the use of indomethacin, have been reported especially for lesions that are not accessible or when there is a danger for cosmetic defect3. In a series of 22 children with cranium EG, we encountered 3 biopsy proven periorbital EGs, in which due to the risk of disfigurement, we administered for a period of 6 months sulfamethoxazole and trimethoprim1. After treatment all lesions were totally resolved. This management has been selected due to previous experience with this regimen in our institute4. Regarding the remaining 19 lesions in our series, radical surgical excision was performed and methylprednisolone was applied in the tumor bed. On follow-up examinations no tumor recurrence was noted1.

We believe that surgery is the option of choice in most lesions of EG that are accessible. For the remaining lesions we propose, apart from intralesional corticosteroid injection, the administration of sulfamethoxazole and trimethoprim as an effective treatment. Future studies should be focused on this issue.


Alexiou A.G., Prodromou N.

Athens. Greece.



1. Alexiou, A.G., Mpairamidis, E., Sfakianos, G., et al.: Cranial unifocal Langerhans cell histiocytosis in children. J Pediatr Surg (in press).        [ Links ]

2. Hoover, K.B., Rosenthal, D.I., Mankin, H.: Langerhans cell histiocytosis. Skeletal Radiol 2007; 36: 95-104.        [ Links ]

3. Munn, S.E., Olliver, L., Broadbent, V., et al.: Use of indomethacin in Langerhans cell histiocytosis. Med Pediatr Oncol 1999; 32: 247-249.        [ Links ]

4. Tzortzatou-Stathopoulou, F., Xaidara, A., Mikraki, V., et al.: Effect of trimethoprim-sulphamethoxazole in langerhans' cell histiocytosis: Preliminary observations. Med Pediatr Oncol 1995; 25: 74-78.        [ Links ]

5. Ulivieri, S., Oliveri, G., Filosomi, G.: Solitary Langerhans cell histiocytosis orbital lesion: case report and review of the literature. Neurocirugía 2008; 19: 453-455.        [ Links ]

Recibido: 9-03-09



Solitary eosinophilc granuloma of bone can present dilemmas of both diagnosis and treatment; since the clinical course of the disease is usually benign, a simple, minimally invasive form of treatment with a low rate of complications is desirable. In view of the possibility of spontaneous healing, complete surgical excision, when it's possible, or biopsy alone to confirm the diagnosis should be a strategy for treatment. My result supports this approach1.



1. Ulivieri, S., Oliveri, G., Filosomi, G.: Solitary Langerhans cell histiocytosis orbital lesion: case report and review of the literature. Neurocirugía. 2008 Oct; 19: 453-455.

S. Ulivieri

Siena. Italia

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