Successful reduction of ACTH secretion in a case of intractable Cushing’s disease with pituitary Crooke’s cell adenoma by combined modality therapy including temozolomide
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- Tanaka Sayuri
- Internal Medicine 1, Shimane University Faculty of Medicine, Izumo 693-8501, Japan
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- Yamamoto Masahiro
- Internal Medicine 1, Shimane University Faculty of Medicine, Izumo 693-8501, Japan
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- Morita Miwa
- Internal Medicine 1, Shimane University Faculty of Medicine, Izumo 693-8501, Japan
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- Takeno Ayumu
- Internal Medicine 1, Shimane University Faculty of Medicine, Izumo 693-8501, Japan
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- Kanazawa Ippei
- Internal Medicine 1, Shimane University Faculty of Medicine, Izumo 693-8501, Japan
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- Yamaguchi Toru
- Internal Medicine 1, Shimane University Faculty of Medicine, Izumo 693-8501, Japan
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- Yamada Shozo
- Endocrine Center, Division of Hypothalamic & Pituitary Surgery, Toranomon Hospital, Tokyo 105-8470, Japan
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- Inoshita Naoko
- Department of Pathology, Toranomon Hospital, Tokyo 105-8470, Japan
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- Oki Yutaka
- Second Division Department of Internal Medicine, Hamamatsu University School of Medicine, Hamamatsu 431-3192, Japan
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- Kurosaki Masamichi
- Division of Neurosurgery, Department of Brain and Neurosciences, Faculty of Medicine Tottori University, Yonago 683-8504, Japan
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- Sugimoto Toshitsugu
- Internal Medicine 1, Shimane University Faculty of Medicine, Izumo 693-8501, Japan
Abstract
<p>Crooke’s cell adenoma (CCA) is an aggressive subtype of corticotroph adenoma; however, CCA is associated with a high incidence of low expression of methyl guanine methyl transferase (MGMT), suggesting that temozolomide (TMZ) treatment might be effective for this tumor type. The case of a 56-year-old woman with Cushing’s disease caused by a pituitary CCA is presented. At the age of 38 years, the patient presented to our hospital with polyuria and a visual field defect. MRI and laboratory studies showed a 4.5-cm-diameter pituitary tumor with plasma adrenocorticotropic hormone (ACTH) and serum cortisol levels of more than 500 pg/mL and 40 μg/dL, respectively. At 39 years of age, the patient underwent a craniotomy, and her plasma ACTH and cortisol levels decreased to less than 200 pg/mL and 10 μg/dL, respectively; however, these hormone levels increased gradually to 3,940 pg/mL and 70 μg/dL, respectively, by the time the patient was 56 years old. Histopathological re-examination of the previously resected specimen showed that the pituitary tumor was MGMT-negative CCA. TMZ treatment after the second operation decreased the plasma ACTH levels from 600–800 pg/mL to 70–300 pg/mL. No signs of recurrence were observed in the seven years following these treatments with added prophylactic radiation therapy. These clinical findings suggest that TMZ treatment to patients with CCA accompanied with elevated ACTH may be good indication to induce lowering ACTH levels and tumor shrinkage.</p>
Journal
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- Endocrine Journal
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Endocrine Journal 66 (8), 701-708, 2019
The Japan Endocrine Society
