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Annals of Oncology Advance Access published online on October 27, 2009
Annals of Oncology, doi:10.1093/annonc/mdp455
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© The Author 2009. Published by Oxford University Press on behalf of the European Society for Medical Oncology. All rights reserved. For permissions, please email: journals.permissions@oxfordjournals.org

MACOP-B regimen in the treatment of adult Langerhans cell histiocytosis: experience on seven patients

E. Derenzini1,*, M. P. Fina1, V. Stefoni1, C. Pellegrini1, F. Venturini1, A. Broccoli1, L. Gandolfi1, S. Pileri2, S. Fanti3, E. Lopci3, P. Castellucci3, C. Agostinelli2, M. Baccarani1 and P. L. Zinzani1

1 Department of Haematology and Medical Oncology ‘L. & A. Seràgnoli’
2 Unit of Haematopathology, Department of Haematology and Medical Oncology ‘L. & A. Seràgnoli’, University of Bologna
3 Department of Nuclear Medicine, Sant'Orsola-Malpighi Hospital, Bologna, Italy

* Correspondence to: Dr E. Derenzini, Department of Haematology and Medical Oncology ‘L. & A. Seràgnoli’, University of Bologna, Via Massarenti 9, 40138 Bologna, Italy. Tel: +39-0516363680; Fax: +39-0516364037; E-mail: enrico.derenzini{at}libero.it

Background: Adult Langerhans cell histiocytosis (LCH) is a rare disease. The combination of vinblastine and prednisone, given in a 6-month course, is the standard of care but prospective randomized trials are lacking.

Patients and methods: We report our monocentric experience in the treatment of seven adult patients with multisystem (MS) LCH (n = 3) or single-system multifocal (SS-m) LCH (n = 4) with the short-course intensive chemotherapy regimen methotrexate, doxorubicin, cyclophosphamide, vincristine, prednisone and bleomicin (MACOP-B).

Results: The overall response rate was 100% [five complete response (CR), two partial response (PR)]. After a median follow-up of 6.5 years, four patients are in first continuous CR and three patients relapsed after 5, 8 and 62 months, respectively. Four patients were evaluated with positron emission tomography (PET) scan: all three PET-negative patients at the end of treatment had a long-lasting response with only one patient relapsing after 5 years. PET scan detected additional bone lesions at diagnosis in two of four patients, changing the treatment program in one of them.

Conclusions: MACOP-B regimen seems to be very active in the treatment of adult MS or SS-m LCH, with long-lasting responses in five of seven patients. PET scan merits further evaluation in the initial staging and in the evaluation of the response to chemotherapy.

chemotherapy, Langerhans cell histiocytosis, MACOP-B

Received for publication April 13, 2009. Revision received July 22, 2009. Accepted for publication August 11, 2009.


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