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Treatment of Nelson's syndrome with temozolomide

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.