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DOI: 10.1530/EJE-08-0557
European Journal of Endocrinology, Vol 160, Issue 1, 115-119
Copyright © 2009 by European Society of Endocrinology
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CASE REPORT

Treatment of Nelson's syndrome with temozolomide

V J Moyes1, G Alusi2, H I Sabin3, J Evanson4, D M Berney5, K Kovacs7, J P Monson1, P N Plowman6 and W M Drake1

Departments of1 Endocrinology2 Otolaryngology3 Neurosurgery, 4 Neuroradiology, 5 , Histopathology6 Oncology, St Bartholomew's Hospital, London EC1A 7BE, UK7 Department of Pathology, St Michael's Hospital, Toronto, M5B 1W8, Canada

(Correspondence should be addressed to W M Drake; Email: w.m.drake{at}qmul.ac.uk)

Abstract

A 64-year-old woman was previously treated for Cushing's disease with trans-sphenoidal surgery, external beam radiotherapy and bilateral adrenalectomy. Progression of an aggressive corticotroph adenoma was evident 3 years post-adrenalectomy; involvement of the clivus was treated with surgery and gamma knife radiosurgery. Tumour spread through the skull base, occiput and left ear with persistent facial pain and left ear discharge; progression continued despite second gamma knife treatment. ACTH levels peaked at 2472 and 2265 pmol/l pre- and post-hydrocortisone respectively. Treatment with temozolomide resulted in a significant improvement in symptoms, a reduction of plasma ACTH to 389 pmol/l and regression of tumour on magnetic resonance imaging scan after four cycles of treatment. We propose that temozolomide is an effective and well-tolerated therapeutic tool for the treatment of Nelson's syndrome and a useful addition to the range of therapies available to treat this condition.




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