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 Blood, 1 November 2005, Vol. 106, No. 9, pp. 2992-2994.
Prepublished online as a Blood First Edition Paper on July 14, 2005; DOI 10.1182/blood-2005-06-2238.

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Submitted June 3, 2005
Accepted June 17, 2005

Successful treatment of Erdheim-Chester disease,a non-Langerhans cell histiocytosis, with interferon-alpha

Fadi Braiteh, Cynthia Boxrud, Bita Esmaeli, and Razelle Kurzrock*

Phase I Program, Division of Cancer Medicine and the University of Texas Graduate School of Biomedical Sciences, Houston, Texas, USA
Jules Stein Eye Institute, UCLA, Santa Monica, California, USA
Section of Ophthalmology, Department of Plastic Surgery, MD Anderson Cancer Center, Houston, Texas, USA

* Corresponding author; email: rkurzroc{at}mdanderson.org.

Erdheim-Chester disease is a rare non-Langerhans histiocytosis with multisystem involvement. To date, there is no standard treatment for this disorder, and over half of the patients succumb within three years. Because interferon-alpha promotes the terminal differentiation of histiocytes and dendritic cells, we hypothesized that this molecule would be a useful therapy for Erdheim-Chester disease. We therefore treated three patients with advanced disease with interferon-alpha at a starting dose of 3 to 6 x106 units, which was later reduced, during maintenance, to 1 x106 units s.c. three times per week. Marked improvement was noted in all patients, with substantial retro-orbital disease regression within one month. Improvement in bone lesions, pain, diabetes insipidus and other manifestations was gradual over many months. Responses were durable (3+ to 4.5+ years). Our observations suggest that this well-tolerated therapy has a significant impact on the course and outcome of Erdheim-Chester disease.


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Interferon-{alpha}: still useful after all these years
Eric J. Feldman
Blood 2005 106: 2930-2931. [Full Text] [PDF]



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