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Interferon alfa-2a maintenance after salvage autologous stem cell transplantation in atypical mycosis fungoides with central nervous system involvement


Doerschner, Miriam; Pekar-Lukacs, Agnes; Messerli-Odermatt, Olivia; Dommann-Scherrer, Corina; Rütti, Markus; Müller, Antonia M; Nair, Gayathri; Kamarachev, Jivko; Kerl, Katrin; Beer, Marc; Messerli, Michael; Frauenknecht, Katrin; Haralambieva, Eugenia; Hoetzenecker, Wolfram; French, Lars E; Guenova, Emmanuella (2018). Interferon alfa-2a maintenance after salvage autologous stem cell transplantation in atypical mycosis fungoides with central nervous system involvement. British Journal of Dermatology:Epub ahead of print.

Abstract

Mycosis fungoides is a primary cutaneous T-cell lymphoma with unfavorable prognosis for the advanced stages of the disease. Refractory disease and advanced-stage disease require systemic therapy. We report on a rare case of an atypical predominantly CD8+ folliculotropic mycosis fungoides (MF), a subtype of MF with poorer prognosis, in a 59-year-old woman, initially diagnosed with MF restricted to the skin- of T3N0M0B0/stage IIB according to the current WHO/EORTC classification. First-line treatment with local percutaneous radiotherapy in combination with systemic interferon alfa-2a resulted in complete remission. However, 21 months later the disease progressed to T3N0M1B0/stage IVB with development of cerebral manifestation and thus very poor prognosis. Allogeneic stem cell transplantation (SCT) was not a therapeutic option due to the lack of a suitable donor. We initiated methotrexate and cytarabin chemotherapy, followed by high-dose chemotherapy with thiotepa and BCNU with autologous SCT. Despite rapid response and complete remission of the cerebral lesions, disease recurrence of the skin occurred soon after. Interestingly, re-administration of interferon alfa-2a as a maintenance treatment after the salvage autologous SCT resulted in a durable complete remission during the follow-up period of currently 13 months after autologous SCT.

Abstract

Mycosis fungoides is a primary cutaneous T-cell lymphoma with unfavorable prognosis for the advanced stages of the disease. Refractory disease and advanced-stage disease require systemic therapy. We report on a rare case of an atypical predominantly CD8+ folliculotropic mycosis fungoides (MF), a subtype of MF with poorer prognosis, in a 59-year-old woman, initially diagnosed with MF restricted to the skin- of T3N0M0B0/stage IIB according to the current WHO/EORTC classification. First-line treatment with local percutaneous radiotherapy in combination with systemic interferon alfa-2a resulted in complete remission. However, 21 months later the disease progressed to T3N0M1B0/stage IVB with development of cerebral manifestation and thus very poor prognosis. Allogeneic stem cell transplantation (SCT) was not a therapeutic option due to the lack of a suitable donor. We initiated methotrexate and cytarabin chemotherapy, followed by high-dose chemotherapy with thiotepa and BCNU with autologous SCT. Despite rapid response and complete remission of the cerebral lesions, disease recurrence of the skin occurred soon after. Interestingly, re-administration of interferon alfa-2a as a maintenance treatment after the salvage autologous SCT resulted in a durable complete remission during the follow-up period of currently 13 months after autologous SCT.

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Additional indexing

Item Type:Journal Article, refereed, original work
Communities & Collections:04 Faculty of Medicine > University Hospital Zurich > Clinic for Hematology
04 Faculty of Medicine > University Hospital Zurich > Institute of Pathology and Molecular Pathology
04 Faculty of Medicine > University Hospital Zurich > Dermatology Clinic
04 Faculty of Medicine > University Hospital Zurich > Clinic for Nuclear Medicine
04 Faculty of Medicine > University Hospital Zurich > Institute of Neuropathology
Dewey Decimal Classification:610 Medicine & health
Uncontrolled Keywords:Dermatology
Language:English
Date:18 December 2018
Deposited On:04 Jan 2019 11:17
Last Modified:29 Apr 2019 07:00
Publisher:Wiley-Blackwell Publishing, Inc.
ISSN:0007-0963
Additional Information:This is the peer reviewed version of the following article: J Eur Acad Dermatol Venereol. 2018 Nov 23. doi: 10.1111/jdv.15361., which has been published in final form at https://doi.org/10.1111/jdv.15361. This article may be used for non-commercial purposes in accordance with Wiley Terms and Conditions for Self-Archiving (http://olabout.wiley.com/WileyCDA/Section/id-820227.html#terms).
OA Status:Closed
Publisher DOI:https://doi.org/10.1111/bjd.17535
PubMed ID:30565216

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