An Adrenocortical Carcinoma Associated with Non-Islet Cell Tumor Hypoglycemia and Aberrant ACTH Production
Introduction. Adrenocortical carcinomas (ACCs) are infrequently reported to present with severe hypoglycemia syndrome resulting from the secretion of insulin-like growth factor II (IGF-II) by tumor cells. Adrenocorticotropic hormone- (ACTH) independent hypercortisolism is the norm of hormonally acti...
Saved in:
| Main Authors: | , , , , , |
|---|---|
| Format: | Article |
| Language: | English |
| Published: |
Wiley
2020-01-01
|
| Series: | Case Reports in Endocrinology |
| Online Access: | http://dx.doi.org/10.1155/2020/2025631 |
| Tags: |
Add Tag
No Tags, Be the first to tag this record!
|
| _version_ | 1832567464830959616 |
|---|---|
| author | M. D. S. A. Dilrukshi A. W. Wickramarachchi D. D. K. Abeyaratne Brian Shine Bahram Jafar-Mohammadi N. P. Somasundaram |
| author_facet | M. D. S. A. Dilrukshi A. W. Wickramarachchi D. D. K. Abeyaratne Brian Shine Bahram Jafar-Mohammadi N. P. Somasundaram |
| author_sort | M. D. S. A. Dilrukshi |
| collection | DOAJ |
| description | Introduction. Adrenocortical carcinomas (ACCs) are infrequently reported to present with severe hypoglycemia syndrome resulting from the secretion of insulin-like growth factor II (IGF-II) by tumor cells. Adrenocorticotropic hormone- (ACTH) independent hypercortisolism is the norm of hormonally active ACCs, but aberrant ACTH production by tumor cells can theoretically cause ACTH-dependent hypercortisolism. The purpose of this report was to present a case of an ACC manifested with the co-occurrence of two extremely rare presentations. Case Description. We present a rare case of a 43-year-old male patient admitted with recurrent episodes of severe non-ketotic and non-insulin-mediated hypoglycemia due to IGF-II mediated disease and ACTH-dependent Cushing’s syndrome. He was diagnosed with a diffusely disseminated adrenocortical carcinoma with immunohistochemistry of tumor cells showing focal ACTH immunostain positivity. Conclusion. Non-islet cell tumor hypoglycemia and ACTH-dependent Cushing’s syndrome are extremely rare presentations of an ACC, and co-occurrence of these entities in a single patient is never reported in the literature. |
| format | Article |
| id | doaj-art-9f218d6b75ce41a7877359b174714b92 |
| institution | Kabale University |
| issn | 2090-6501 2090-651X |
| language | English |
| publishDate | 2020-01-01 |
| publisher | Wiley |
| record_format | Article |
| series | Case Reports in Endocrinology |
| spelling | doaj-art-9f218d6b75ce41a7877359b174714b922025-02-03T01:01:26ZengWileyCase Reports in Endocrinology2090-65012090-651X2020-01-01202010.1155/2020/20256312025631An Adrenocortical Carcinoma Associated with Non-Islet Cell Tumor Hypoglycemia and Aberrant ACTH ProductionM. D. S. A. Dilrukshi0A. W. Wickramarachchi1D. D. K. Abeyaratne2Brian Shine3Bahram Jafar-Mohammadi4N. P. Somasundaram5Diabetes and Endocrinology Unit of National Hospital of Sri Lanka, Colombo 10, Sri LankaNational Hospital of Sri Lanka, Colombo, Sri LankaDiabetes and Endocrinology Unit of National Hospital of Sri Lanka, Colombo 10, Sri LankaClinical Biochemistry, John Radcliffe Hospital, Oxford OX3 9DU, UKOxford Centre for Diabetes, Endocrinology and Metabolism, Churchill Hospital, Oxford University Hospitals NHS Foundation Trust, Oxford, UKDiabetes and Endocrinology Unit of National Hospital of Sri Lanka, Colombo 10, Sri LankaIntroduction. Adrenocortical carcinomas (ACCs) are infrequently reported to present with severe hypoglycemia syndrome resulting from the secretion of insulin-like growth factor II (IGF-II) by tumor cells. Adrenocorticotropic hormone- (ACTH) independent hypercortisolism is the norm of hormonally active ACCs, but aberrant ACTH production by tumor cells can theoretically cause ACTH-dependent hypercortisolism. The purpose of this report was to present a case of an ACC manifested with the co-occurrence of two extremely rare presentations. Case Description. We present a rare case of a 43-year-old male patient admitted with recurrent episodes of severe non-ketotic and non-insulin-mediated hypoglycemia due to IGF-II mediated disease and ACTH-dependent Cushing’s syndrome. He was diagnosed with a diffusely disseminated adrenocortical carcinoma with immunohistochemistry of tumor cells showing focal ACTH immunostain positivity. Conclusion. Non-islet cell tumor hypoglycemia and ACTH-dependent Cushing’s syndrome are extremely rare presentations of an ACC, and co-occurrence of these entities in a single patient is never reported in the literature.http://dx.doi.org/10.1155/2020/2025631 |
| spellingShingle | M. D. S. A. Dilrukshi A. W. Wickramarachchi D. D. K. Abeyaratne Brian Shine Bahram Jafar-Mohammadi N. P. Somasundaram An Adrenocortical Carcinoma Associated with Non-Islet Cell Tumor Hypoglycemia and Aberrant ACTH Production Case Reports in Endocrinology |
| title | An Adrenocortical Carcinoma Associated with Non-Islet Cell Tumor Hypoglycemia and Aberrant ACTH Production |
| title_full | An Adrenocortical Carcinoma Associated with Non-Islet Cell Tumor Hypoglycemia and Aberrant ACTH Production |
| title_fullStr | An Adrenocortical Carcinoma Associated with Non-Islet Cell Tumor Hypoglycemia and Aberrant ACTH Production |
| title_full_unstemmed | An Adrenocortical Carcinoma Associated with Non-Islet Cell Tumor Hypoglycemia and Aberrant ACTH Production |
| title_short | An Adrenocortical Carcinoma Associated with Non-Islet Cell Tumor Hypoglycemia and Aberrant ACTH Production |
| title_sort | adrenocortical carcinoma associated with non islet cell tumor hypoglycemia and aberrant acth production |
| url | http://dx.doi.org/10.1155/2020/2025631 |
| work_keys_str_mv | AT mdsadilrukshi anadrenocorticalcarcinomaassociatedwithnonisletcelltumorhypoglycemiaandaberrantacthproduction AT awwickramarachchi anadrenocorticalcarcinomaassociatedwithnonisletcelltumorhypoglycemiaandaberrantacthproduction AT ddkabeyaratne anadrenocorticalcarcinomaassociatedwithnonisletcelltumorhypoglycemiaandaberrantacthproduction AT brianshine anadrenocorticalcarcinomaassociatedwithnonisletcelltumorhypoglycemiaandaberrantacthproduction AT bahramjafarmohammadi anadrenocorticalcarcinomaassociatedwithnonisletcelltumorhypoglycemiaandaberrantacthproduction AT npsomasundaram anadrenocorticalcarcinomaassociatedwithnonisletcelltumorhypoglycemiaandaberrantacthproduction AT mdsadilrukshi adrenocorticalcarcinomaassociatedwithnonisletcelltumorhypoglycemiaandaberrantacthproduction AT awwickramarachchi adrenocorticalcarcinomaassociatedwithnonisletcelltumorhypoglycemiaandaberrantacthproduction AT ddkabeyaratne adrenocorticalcarcinomaassociatedwithnonisletcelltumorhypoglycemiaandaberrantacthproduction AT brianshine adrenocorticalcarcinomaassociatedwithnonisletcelltumorhypoglycemiaandaberrantacthproduction AT bahramjafarmohammadi adrenocorticalcarcinomaassociatedwithnonisletcelltumorhypoglycemiaandaberrantacthproduction AT npsomasundaram adrenocorticalcarcinomaassociatedwithnonisletcelltumorhypoglycemiaandaberrantacthproduction |