Management of non-malignant hyperinsulinemic hypoglycaemia in resource limited setting: a case report
Abstract
A 37 y.o. Asian male presented with frequent hypoglycaemia in both fasting and post-prandial state. He had elevated blood insulin levels during the hypoglycaemic episodes with normal pancreatic morphology and no extra-pancreatic tumor. Through systematic symptom assessment and the use of simple laboratory examination, non-insulinoma pancreatogenous hypoglycaemia syndrome presented as the most likely diagnosis. Conclusive aetiological diagnosis could not be reached due to the limited availability of diagnostic modalities. Nevertheless, modest symptom control and frequency reduction of hypoglycaemia were achieved with empirical dietary modification and α-glucosidase inhibitor treatment.
References
Kapoor RR, James C, Hussain K. Advances in the diagnosis and management of hyperinsulinemic hypoglycemia. Nat Clin Pract Endocrinol Metab 2009; 5(2):101-12.
https://doi.org/10.1038/ncpendmet1046
Cappellani D, Macchia E, Falorni A, Marchetti P. Insulin Autoimmune Syndrome (Hirata Disease): A Comprehensive Review Fifty Years After Its First Description. Diabetes Metab Syndr Obes 2020; 13:963-78.
https://doi.org/0.2147/DMSO.S219438
Nadelson J, Epstein A. A rare case of noninsulinoma pancreatogenous hypoglycemia syndrome. Case Rep Gastrointest Med 2012; 2012:164305.
https://doi.org/10.1155/2012/164305
Chen F, Yang J, Liu Y, Wang W, Zhu L, Wang W, et al. Insulin autoimmune syndrome: Three case reports. Medicine (Baltimore) 2018; 97(51):e13486.
https://doi.org/10.1097/MD.0000000000013486
Douillard C, Jannin A, Vantyghem MC. Rare causes of hypoglycemia in adults. Ann Endocrinol (Paris) 2020; 81(2-3):110-7.
https://doi.org/10.1016/j.ando.2020.04.003
Okabayashi T, Shima Y, Sumiyoshi T, Kozuki A, Ito S, Ogawa Y, et al. Diagnosis and management of insulinoma. World J Gastroenterol 2013; 19(6):829-37.
https://doi.org/10.3748/wjg.v19.i6.829
Church D, Cardoso L, Kay RG, Williams CL, Freudenthal B, Clarke C, et al. Assessment and Management of Anti-Insulin Autoantibodies in Varying Presentations of Insulin Autoimmune Syndrome. J Clin Endocrinol Metab 2018; 103(10):3845-55.
https://doi.org/10.1210/jc.2018-00972
Service FJ, Natt N, Thompson GB, Grant CS, van Heerden JA, Andrews JC, et al. Noninsulinoma pancreatogenous hypoglycemia: a novel syndrome of hyperinsulinemic hypoglycemia in adults independent of mutations in Kir6.2 and SUR1 genes. J Clin Endocrinol Metab 1999; 84(5):1582-9.
https://doi.org/10.1210/jcem.84.5.5645
Thompson GB, Service FJ, Andrews JC, Lloyd RV, Natt N, van Heerden JA, et al. Noninsulinoma pancreatogenous hypoglycemia syndrome: an update in 10 surgically treated patients. Surgery 2000; 128(6):937-44;discussion 44-5.
https://doi.org/10.1067/msy.2000.110243
Thompson SM, Vella A, Thompson GB, Rumilla KM, Service FJ, Grant CS, et al. Selective Arterial Calcium Stimulation With Hepatic Venous Sampling Differentiates Insulinoma From Nesidioblastosis. J Clin Endocrinol Metab 2015; 100(11):4189-97.
https://doi.org/10.1210/jc.2015-2404
Schwetz V, Horvath K, Kump P, Lackner C, Perren A, Forrer F, et al. Successful Medical Treatment of Adult Nesidioblastosis With Pasireotide over 3 Years: A Case Report. Medicine (Baltimore) 2016; 95(14):e3272.
https://doi.org/10.1097/MD.0000000000003272
Nadelson J, Epstein A. A rare case of noninsulinoma pancreatogenous hypoglycemia syndrome. Case Rep Gastrointest Med 2012; 2012:164305.
https://doi.org/10.1155/2012/164305
Hayashida R, Tsuchiya K, Sekine T, Momose T, Sato F, Sakurada M, et al. A Clinical Case of Insulinoma Presenting with Postprandial Hypoglycemia in a Patient with a History of Gastric Bypass Surgery. Intern Med 2022; 61(8):1189-95.
https://doi.org/10.2169/internalmedicine.7428-21
Bernard B, Kline GA, Service FJ. Hypoglycaemia following upper gastrointestinal surgery: case report and review of the literature. BMC Gastroenterol 2010; 10:77.
https://doi.org/10.1186/1471-230X-10-77
Anderson B, Nostedt J, Girgis S, Dixon T, Agrawal V, Wiebe E, et al. Insulinoma or non-insulinoma pancreatogenous hypoglycemia? A diagnostic dilemma. J Surg Case Rep 2016; 2016(11):rjw188.
https://doi.org/10.1093/jscr/rjw188
Tsujino M, Sugiyama T, Nishida K, Takada Y, Takanishi K, Ishizawa M, et al. Noninsulinoma pancreatogenous hypoglycemia syndrome: a rare case of adult-onset nesidioblastosis. Intern Med 2005; 44(8):843-7.
https://doi.org/10.2169/internalmedicine.44.843
Preechasuk L, Pongpaibul A, Kunavisarut T. Non-insulinoma Pancreatogeneous Hypoglycemia Syndrome with False-Positive Somatostatin Receptor Scintigraphy: A Case Report and Review of Literature. J Med Assoc Thai 2016; 99(3):354-9.
Kellogg TA, Bantle JP, Leslie DB, Redmond JB, Slusarek B, Swan T, et al. Postgastric bypass hyperinsulinemic hypoglycemia syndrome: characterization and response to a modified diet. Surg Obes Relat Dis 2008; 4(4):492-9.
https://doi.org/10.1016/j.soard.2008.05.005
DiNicolantonio JJ, Bhutani J, O'Keefe JH. Acarbose: safe and effective for lowering postprandial hyperglycaemia and improving cardiovascular outcomes. Open Heart 2015; 2(1):e000327.
https://doi.org/10.1136/openhrt-2015-000327
Chen X, Feng L, Yao H, Yang L, Qin Y. Efficacy and safety of diazoxide for treating hyperinsulinemic hypoglycemia: A systematic review and meta-analysis. PLoS One 2021; 16(2):e0246463.
https://doi.org/10.1371/journal.pone.0246463
Haris B, Saraswathi S, Hussain K. Somatostatin analogues for the treatment of hyperinsulinaemic hypoglycaemia. Ther Adv Endocrinol Metab 2020; 11:2042018820965068.
https://doi.org/10.1177/2042018820965068
Guseva N, Phillips D, Mordes JP. Successful treatment of persistent hyperinsulinemic hypoglycemia with nifedipine in an adult patient. Endocr Pract 2010; 16(1):107-11.
