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© 1996 British Society for Rheumatology

case-report

SEVERE OSTEOPOROSIS DUE TO SYSTEMIC MAST CELL DISEASE: SUCCESSFUL TREATMENT WITH INTERFERON ALPHA-2B

T. LEHMANN, C. BEYELER, B. LÄMMLE*, T. HUNZIKER{dagger}, P. VOCK{ddagger}, A. J. OLAH§, C. DAHINDEN and N. J. GERBER

Divisions of Rheumatology 3010 Berne, Switzerland
*Haematology 3010 Berne, Switzerland
{dagger}Dermatology 3010 Berne, Switzerland
{ddagger}Diagnostic Radiology 3010 Berne, Switzerland
§Anatomy 3010 Berne, Switzerland
¶Immunology, University Hospital 3010 Berne, Switzerland

Correspondence to: Correspondence to: Christine Beyeler, Department of Rheumatology, University Hospital, CH-3010 Beme, Switzerland

We describe a 33-yr-old man suffering from severe vertebral osteoporosis and urticaria pigmentosa due to systemic mast cell disease (SMCD). Because i.v. clodronate therapy could not prevent further vertebral fractures, an additional treatment with interferon alpha-2b was initiated. During 24 months of treatment, our patient had no further pain episodes, no new vertebral fractures were discovered, trabecular bone mineral density (BMD) increased significantly and urticarial symptoms improved. Nevertheless, the extent of skin lesions remained unchanged. On histological examination, a remarkable decrease of mast cells was observed in the bone marrow, but not in the skin. Five months after discontinuation of interferon alpha-2b, trabecular BMD decreased and urticarial symptoms deteriorated. These findings illustrate a beneficial effect of interferon alpha-2b on SMCD-induced osteoporosis as well as urticarial symptoms, and raise the question whether this treatment may have a diverse impact on mast cell populations in different tissues.

KEY WORDS: Osteoporosis, Systemic mast cell disease, Interferon alpha


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