Findings that shed new light on the possible pathogenesis of a disease or an adverse effect: Cushing’s syndrome and big IGF-II associated hypoglycaemia in a patient with adrenocortical carcinoma

doi:10.1136/bcr.07.2009.2100

BMJ Case Rep. 2010; 2010: bcr07.2009.2100.

Published online 2010 March 30. doi: 10.1136/bcr.07.2009.2100

PMCID: PMC3027795

Findings that shed new light on the possible pathogenesis of a disease or an adverse effect

Cushing’s syndrome and big IGF-II associated hypoglycaemia in a patient with adrenocortical carcinoma

Kazuhide Ishikura,¹ Toshinari Takamura,¹ Yumie Takeshita,¹ Atsushi Nakagawa,² Noriko Imaizumi,² Hirofumi Misu,¹ Komei Taji,¹ Kazuo Kasahara,³ Yukinosuke Oshinoya,⁴ Shioto Suzuki,⁵ Akishi Ooi,⁵ and Shuichi Kaneko¹

¹Kanazawa University Graduate School of Medical Science, Department of Disease Control and Homeostasis, 13-1 Takara-machi, Kanazawa, Ishikawa, 920-8641, Japan

²Kanazawa Medical University, Department of Internal Medicine, Division of Endocrinology and Metabolism, 1-1 Daigaku, Uchinada, Ishikawa, 920-0293, Japan

³Kanazawa University Graduate School of Medical Science, Respiratory Medicine, 13-1 Takara-machi, Kanazawa, Ishikawa, Ishikawa, 920-8641, Japan

⁴Public Central Hospital of Matto Ishikawa, Urology, 3-8 Kuramitsu, Hakusan, Ishikawa, 924-0865, Japan

⁵Kanazawa University Graduate School of Medical Science, Department of Molecular and Cellular Pathology, 13-1 Takara-machi, Kanazawa, Ishikawa, 920-8641, Japan

Correspondence to Toshinari Takamura, Email: ttakamura/at/m-kanazawa.jp

Abstract

A 41-year-old woman had a general health examination and was diagnosed with a non-functioning adrenocortical carcinoma (ACC). Despite surgery and chemotherapy with mitotane, the ACC progressed with metastases to the lymph nodes, liver and lung. Initially, she developed adrenal insufficiency and was treated with hydrocortisone. As the ACC progressed, it produced superabundant cortisol, resulting in clinically overt Cushing’s syndrome. As the liver metastases grew, the patient developed hypoglycaemia with suppression of endogenous insulin secretion. She had to be given large quantities of glucose intravenously to remain normoglycaemic. The serum insulin-like growth factor (IGF)-II/IGF-I ratio had increased to 84. We identified big IGF-II, a primary hormonal mediator of non-islet cell tumour hypoglycaemia (NICTH), in the serum and tumour using western blotting. This is the first case of ACC that showed both Cushing’s syndrome and NICTH associated with big IGF-II.

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