Tumor-Induced Hypoglycemia: An Unusual Case Report and Review of Literature

Abdulhadi, Basma; Anastasopoulou, Catherine; Lekprasert, Patamaporn

doi:10.1016/j.aace.2020.11.002

Cited by 7 publications

(9 citation statements)

References 19 publications

(3 reference statements)

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“…Therefore, keeping hypoglycemia in mind as a possible finding in such patients, such patients presenting with severe hypoglycemia should be ruled out for nonsolid tumors or hematological diseases to manage them on time and save their lives. For example, one may plan to conduct an initial evaluation to determine the cause of hypoglycemia by taking a detailed history to find out the nature of symptoms and how these are related to time, for example, is it related to a meal or not [ 20 ]. Furthermore, the physician should also evaluate the patient for any signs and symptoms of the tumor as well as medications taken by the patient.…”

Section: Discussionmentioning

confidence: 99%

Rare Presentation of Hypoglycemia in a Patient with Anaplastic Large-Cell Lymphoma

Aldahash

2021

Case Reports in Endocrinology

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Anaplastic large-cell lymphoma is a rare type of aggressive non-Hodgkin’s lymphoma, and arriving at a final diagnosis for this tumor is a challenge for the healthcare providers. Usually, it involves the lymph nodes and extranodal tissues such as the lungs, skin, and other soft tissues. Its presentation by extending into different organs such as the liver, lungs, bones, spleen, and thyroid is rare. Thus, involvement of other organs is very rare as we found in a 54-year-old male patient, a known case of hypertension and end-stage renal disease who was on hemodialysis, who presented to the emergency department with a history of generalized weakness and weight loss of about 20 kg for two months. The tumor cells are positive for CD45, CD30, CD15, MUMi, and Ki-67 (80%) and negative for CD20, PAX-5, CD79a, CD3, CD5, CD10, BCL6, BCL2, EMA, ALK-1, and CD138. The patient was hypoglycemic and hypercalcemic and was managed accordingly. The patient was evaluated, and the third assessment showed that hypoglycemia was resolved due to dexamethasone. The patient’s glucose storage was depleted most likely due to liver involvement plus poor general condition. It was asserted that the patient’s hypoglycemia could be related to his underlying malignancy. Also, the patient was advised to start tablet diazoxide 45 mg three times a day (3 mg/kg/day TID) in addition to levothyroxine tablet 50 mcg once a day. Tablet diazoxide was stopped, and nutritional support was recommended. This case reveals a rare systematic ALK-1-negative anaplastic large-cell lymphoma that involves multiple organs. The main learning point from this report is that these tumors can present atypically even in adults and can be ALK-1 negative, which is contrary to the typical systematic anaplastic large-cell lymphomas that are positive for ALK.

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Section: Discussionmentioning

confidence: 99%

Rare Presentation of Hypoglycemia in a Patient with Anaplastic Large-Cell Lymphoma

Aldahash

2021

Case Reports in Endocrinology

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“…Hyperglycaemia is common and is probably caused by two effects of catecholamines, namely increased insulin resistance and inhibition of insulin secretion from beta cells (14). Hypoglycaemic attacks are rare, only a few cases have been reported (15,16,17,18) and the mechanism is not known. It has been postulated (16) that catecholamine induced β-adrenoceptor-mediated release of insulin from pancreatic β-cells may override the prevailing α-adrenoceptor mediated inhibition of release and the insulin resistance seen in most phaeochromocytoma patients (14).…”

Section: Expression Of Ins-igf2 Transcript Analysed As In (A and C) S...mentioning

confidence: 99%

“…In addition, we suggest that insulin-related molecules partly mimicking insulin may block the insulin receptor, and thereby contribute to hyperglycaemia. Secondly, rare cases with hypoglycaemic attacks have been reported in phaeochromocytoma patients (15,16,17,18) but the mechanism is unknown. Hypersecretion of insulin from the tumour may cause hypoglycaemia.…”

Section: Introductionmentioning

confidence: 99%

Phaeochromocytomas overexpress insulin transcript and produce insulin

Folling¹,

Wennerstrom²,

Eide³

et al. 2022

EJEA

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Introduction: Phaeochromocytomas are tumours originating in the medulla of the adrenal gland. They produce catecholamines, and some tumours also produce ectopic hormones. Two types of glucose imbalances occur in phaeochromocytoma patients, hyperglycaemia and hypoglycaemic attacks. Therefore, we tested whether insulin transcript (INS), insulin, and a hybrid read-through transcript between exons from insulin and insulin-like growth factor 2 (INS-IGF2) were expressed in phaeochromocytomas. Methods: We measured the expression of insulin using immunohistochemistry. The expression of INS-IGF2 was determined by qRT-PCR in formalin-fixed and paraffinembedded tissue from 20 phaeochromocytomas. The expression of INS and INS-IGF2 transcripts was also analysed in 182 phaeochromocytomas and paragangliomas using publicly available datasets in The Cancer Genome Atlas (TCGA) Database. Results: Of 20 phaeochromocytomas, 16 stained positive for insulin. The distribution of positive cells was mostly scattered, with some focal expression indicating clonal expansion. Nineteen tumours expressed high levels of INS and INS-IGF2 transcripts. The expression of the two transcripts corresponded closely. In the TCGA dataset, phaeochromocytoma expresses higher levels of INS and INS-IGF2 transcripts compared to the normal non-tumour adrenal glands. Thus, the expression of INS and INS-IGF2 seems to be a general phenomenon in phaeochromocytoma. Conclusion: Most phaeochromocytomas contain cells that overexpress INS and INS-IGF2 transcripts. Most tumours also display heterogeneous expression of polypeptides immunoreactive to monoclonal anti-insulin antibodies. Clinically this may relate to both hyperglycaemia and hypoglycaemic attacks seen in patients with phaeochromocytoma as well as autocrine tumour growth.

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“…Hyperglycaemia is common, and is probably caused by two effects of catecholamines, namely increased insulin resistance and inhibition of insulin secretion from beta cells (14). Hypoglycaemic attacks are rare, only few cases have been reported (15)(16)(17)(18) and the mechanism is not known. It has been postulated ( 16) that catecholamine induced β-adrenoceptor mediated release of insulin from pancreatic β-cells may override the prevailing α-adrenoceptor mediated inhibition of release and the insulin resistance seen in most phaeochromocytoma patients (14).…”

Section: Several Lines Of Evidence Further Support a Possibility Of Insulin Expression In Phaeochromocytomas;mentioning

confidence: 99%

“…In addition, we suggest that insulin-related molecules partly mimicking insulin may block the insulin receptor, and thereby contribute to hyperglycaemia. Secondly, rare cases with hypoglycaemic attacks have been reported in phaeochromocytoma patients ( 15 , 16 , 17 , 18 ) but the mechanism is unknown. Hypersecretion of insulin from the tumour may cause hypoglycaemia.…”

Section: Introductionmentioning

confidence: 99%

Phaeochromocytomas overexpress insulin transcript and produce insulin

Følling

Wennerstrøm

Eide

et al. 2021

Endocrine Connections

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Introduction: Phaeochromocytomas are tumours originating in the medulla of the adrenal gland. They produce catecholamines and some tumours also produce ectopic hormones. Production of insulin has not been reported. However, two types of glucose imbalances occur in pheaochromocytoma patients, hyperglycaemia and hypoglycaemic attacks. Therefore, we tested whether insulin was expressed in phaeochromocytomas. Methods: We measured the expression of insulin using immunohistochemistry. The expression of insulin transcript (INS) and a hybrid read-through transcript between exons from insulin and insulin-like growth factor 2 (INS-IGF2) was determined by qRT-PCR in formalin-fixed and paraffin-embedded tissue from 20 phaeochromocytomas. The expression of INS and INS-IGF2 transcripts was also analysed in 182 phaeochromocytomas and paragangliomas using publicly available datasets in The Cancer Genome Atlas (TCGA) Database. Results: Of 20 phaeochromocytomas, 16 stained positive for insulin. The distribution of positive cells was mostly scattered, with some focal expression indicating clonal expansion. 19 tumours expressed high levels of INS and INS-IGF2 transcripts. The expression of the two transcripts correlated positively. In the TCGA dataset, phaeochromocytoma express higher levels of INS and INS-IGF2 transcripts compared to normal non-tumour adrenal gland tissue (p=0.01). Thus, expression of INS and INS-IGF2 seems to be a general phenomenon in phaeochromocytoma. Conclusion: Most phaeochromocytomas contain cells that express the INS and INS-IGF2 transcripts. As most tumours also express polypeptides immunoreactive to monoclonal anti-insulin antibodies, expression of the transcripts are likely functionally relevant. Clinically this may relate to both hyperglycaemia and hypoglycaemic attacks seen in patients with a phaeochromocytoma.

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Tumor-Induced Hypoglycemia: An Unusual Case Report and Review of Literature

Cited by 7 publications

References 19 publications

Rare Presentation of Hypoglycemia in a Patient with Anaplastic Large-Cell Lymphoma

Rare Presentation of Hypoglycemia in a Patient with Anaplastic Large-Cell Lymphoma

Phaeochromocytomas overexpress insulin transcript and produce insulin

Phaeochromocytomas overexpress insulin transcript and produce insulin

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