Cushing's syndrome and big IGF-II associated hypoglycaemia in a patient with adrenocortical carcinoma
Open accessAbstract
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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Tumor-associated hypoglycemia is a well-described phenomenon, historically termed Anderson's syndrome, which may be attributed to IGF-2-mediated hypoglycemia.
“However, tumor-associated hypoglycemia is a well-described phenomenon, historically termed Anderson's syndrome, which may be attributed to IGF-2–mediated hypoglycemia (79–81).”
From Adrenocortical carcinoma (2014) · cites this paper as [79]
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Else, T., Kim, A. C., Sabolch, A., Raymond, V. M., Kandathil, A., Caoili, E. M., Jolly, S., Miller, B. S., Giordano, T. J., & Hammer, G. D. (2014) · Endocrine Reviews · doi:10.1210/er.2013-1029 · Cited by 892 · see citing works
- Authors
- Ishikura, K., Takamura, T., Takeshita, Y., Nakagawa, A., Imaizumi, N., Misu, H., Taji, K., Kasahara, K., Oshinoya, Y., Suzuki, S., Ooi, A., & Kaneko, S.
- Affiliations
- Kanazawa University (JP)
- Kanazawa Medical University (JP)
- Public Central Hospital of Matto Ishikawa (JP)
- Corresponding author
- Takamura, Toshinari
- Venue
- BMJ Case Reports
- Publisher
- BMJ
- Published
- 2010
- Volume
- 2010
- Pages
- bcr0720092100
- ISSN
- 1757-790X (ISSN-L)
- PMC
- PMC3027795
- OpenAlex
- W2103219440
- Cited by
- 20
- References
- 17
- Citations by year
- 2025: 2 · 2024: 1 · 2023: 1 · 2022: 2 · 2021: 2 · 2020: 1 · 2019: 1 · 2016: 4 · 2015: 1 · 2014: 1 · 2013: 3 · 2012: 1
- FWCI
- 1.35
- Type
- journal article
- Language
- en
- Open access
- green
- www.ncbi.nlm.nih.gov
- Locations
- BMJ Case Reports (publishedVersion)
- Europe PMC (PubMed Central) (publishedVersion)
- PubMed Central (publishedVersion)
- PubMed (publishedVersion)
- Topics
- Health Sciences › Medicine › Surgery › Adrenal and Paraganglionic Tumors
- Health Sciences › Medicine › Endocrinology, Diabetes and Metabolism › Pituitary Gland Disorders and Treatments
- Health Sciences › Medicine › Epidemiology › Neuroendocrine Tumor Research Advances
- Keywords
- Cushing's syndrome, adrenocortical carcinoma, insulin-like growth factor II, adrenal insufficiency, mitotane, liver metastasis, hypercortisolism
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