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Reactive hypoglycemia owing to an intrahepatic congenital portosystemic shunt in an older patient

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Abstract

An 85-year-old woman was admitted to our hospital because of hypoglycemia and impairment of consciousness several hours after breakfast. Because the hypoglycemia predominantly occurred 2–4 h after meals, we diagnosed reactive hypoglycemia. An oral glucose tolerance test showed prolonged hyperinsulinemia following the postprandial hyperglycemia, with a subsequent rapid decrease in blood glucose concentration. The post-stimulus plasma C-peptide concentration was relatively low compared to the plasma insulin concentration. Abdominal computed tomography revealed an intrahepatic congenital portosystemic shunt (CPSS). On the basis of these findings, we concluded that the reactive hypoglycemia was induced by the CPSS, via a reduction in hepatic insulin extraction. Treatment with an alpha-glucosidase inhibitor resolved the reactive hypoglycemia. CPSS comprises anomalous vascular connections between the portal vein and the systemic venous circulation, and reactive hypoglycemia is a rare complication of this malformation, which has most frequently been reported in children, with only a few cases reported in adults. However, this case indicates that even in adult patients, imaging studies should be conducted to rule out CPSS as the cause of the reactive hyperglycemia.

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Data availability

The data in the present report are not publicly available but are available from the corresponding author on reasonable request.

References

  1. Hofeldt FD. Reactive hypoglycemia. Metabolism. 1975;24(10):1193–208.

    Article  CAS  PubMed  Google Scholar 

  2. Shah P, Rahman SA, Demirbilek H, et al. Hyperinsulinaemic hypoglycaemia in children and adults. Lancet Diabetes Endocrinol. 2017;5(9):729–42.

    Article  CAS  PubMed  Google Scholar 

  3. Tsitouridis I, Sotiriadis C, Michaelides M, et al. Intrahepatic portosystemic venous shunts: radiological evaluation. Diagn Interv Radiol. 2009;15(3):182–7.

    PubMed  Google Scholar 

  4. Palvanov A, Marder RL, Siegel D. Asymptomatic intrahepatic portosystemic venous shunt: to treat or not to treat? Int J Angiol. 2016;25(3):193–8.

    PubMed  Google Scholar 

  5. Pocha C, Maliakkal B. Spontaneous intrahepatic portal-systemic venous shunt in the adult: case report and review of the literature. Dig Dis Sci. 2004;49(7–8):1201–6.

    Article  PubMed  Google Scholar 

  6. Sokollik C, Bandsma RH, Gana JC, et al. Congenital portosystemic shunt: characterization of a multisystem disease. J Pediatr Gastroenterol Nutr. 2013;56(6):675–81.

    Article  PubMed  Google Scholar 

  7. Xu S, Zhang P, Hu L, et al. Case report: clinical features of congenital portosystemic shunts in the neonatal period. Front Pediatr. 2021;9:778–91.

    Article  Google Scholar 

  8. Uchino T, Matsuda I, Endo F. The long-term prognosis of congenital portosystemic venous shunt. J Pediatr. 1999;135(2 Pt 1):254–6.

    Article  CAS  PubMed  Google Scholar 

  9. Paganelli M, Lipsich JE, Sciveres M, et al. Predisposing factors for spontaneous closure of congenital portosystemic shunts. J Pediatr. 2015;167(4):931–5.

    Article  PubMed  Google Scholar 

  10. Peček J, Fister P, Homan M. Abernethy syndrome in Slovenian children: five case reports and review of literature. World J Gastroenterol. 2020;26(37):5731–44.

    Article  PubMed  PubMed Central  Google Scholar 

  11. Bas S, Guran T, Atay Z, et al. Premature pubarche, hyperinsulinemia and hypothyroxinemia: novel manifestations of congenital portosystemic shunts (Abernethy malformation) in children. Horm Res Paediatr. 2015;83(4):282–7.

    Article  CAS  PubMed  Google Scholar 

  12. Senniappan S, Pitt K, Shah P, et al. Postprandial hyperinsulinaemic hypoglycaemia secondary to a congenital portosystemic shunt. Horm Res Paediatr. 2015;83(3):217–20.

    Article  CAS  PubMed  Google Scholar 

  13. Weigert A, Bierwolf J, Reutter H, et al. Congenital intrahepatic portocaval shunts and hypoglycemia due to secondary hyperinsulinism: a case report and review of the literature. J Med Case Rep. 2018;12(1):336.

    Article  PubMed  PubMed Central  Google Scholar 

  14. Yoshii K, Noda M, Naiki Y, et al. Portosystemic shunt as a cause of congenital hyperinsulinemic hypoglycemia. Pediatr Int. 2017;59(4):512–4.

    Article  PubMed  Google Scholar 

  15. Ghasemi-Rad M, Smuclovisky E, Cleveland H, et al. Endovascular treatment of a portosystemic shunt presenting with hypoglycemia; case presentation and review of literature. Clin Imaging. 2022;83:131–7.

    Article  PubMed  Google Scholar 

  16. Duprey J, Gouin B, Benazet MF, et al. Glucose intolerance and post-stimulatory hypoglycemia secondary to a probably congenital intrahepatic portacaval anastomosis. Ann Med Interne (Paris). 1985;136(8):655–8 (Abstract only).

    CAS  PubMed  Google Scholar 

  17. Chagnon SF, Vallee CA, Barge J, et al. Aneurysmal portahepatic venous fistula: report of two cases. Radiology. 1986;159(3):693–5.

    Article  CAS  PubMed  Google Scholar 

  18. Gouin B, Le Gal J, Duprey J, et al. Congenital intrahepatic portacaval anastomosis: analysis of manifested glucose abnormalities. Gastroenterol Clin Biol. 1984;8(5):464–8 (Abstract only).

    CAS  PubMed  Google Scholar 

  19. Bratusch-Marrain PR, Waldhäusl WK, Gasić S, et al. Hepatic disposal of biosynthetic human insulin and porcine C-peptide in humans. Metabolism. 1984;33(2):151–7.

    Article  CAS  PubMed  Google Scholar 

  20. Meier JJ, Veldhuis JD, Butler PC. Pulsatile insulin secretion dictates systemic insulin delivery by regulating hepatic insulin extraction in humans. Diabetes. 2005;54(6):1649–56.

    Article  CAS  PubMed  Google Scholar 

  21. De León DD, Stanley CA. Determination of insulin for the diagnosis of hyperinsulinemic hypoglycemia. Best Pract Res Clin Endocrinol Metab. 2013;27(6):763–9.

    Article  PubMed  PubMed Central  Google Scholar 

  22. Harris MI, Cowie CC, Gu K, et al. Higher fasting insulin but lower fasting C-peptide levels in African Americans in the US population. Diabetes Metab Res Rev. 2002;18(2):149–55.

    Article  CAS  PubMed  Google Scholar 

  23. Faber OK, Christensen K, Kehlet H, et al. Decreased insulin removal contributes to hyperinsulinemia in obesity. J Clin Endocrinol Metab. 1981;53(3):618–21.

    Article  CAS  PubMed  Google Scholar 

  24. Iwasaki Y, Ohkubo A, Kajinuma H, et al. Degradation and secretion of insulin in hepatic cirrhosis. J Clin Endocrinol Metab. 1978;47(4):774–9.

    Article  CAS  PubMed  Google Scholar 

  25. Johnson DG, Alberti KG, Faber OK, et al. Hyperinsulinism of hepatic cirrhosis: diminished degradation or hypersecretion? Lancet. 1977;1(8001):10–3.

    Article  CAS  PubMed  Google Scholar 

  26. Heding LG, Rasmussen SM. Human C-peptide in normal and diabetic subjects. Diabetologia. 1975;11(3):201–6.

    Article  CAS  PubMed  Google Scholar 

  27. Kajinuma H, Kanazawa Y, Sando H, et al. Human plasma C-peptide immunoreactivity: its correlation with immunoreactive insulin in diabetes, and chronic liver and renal diseases. Endocrinol Jpn. 1979;26(1):65–73.

    Article  CAS  PubMed  Google Scholar 

  28. Joshi SR, Standl E, Tong N, et al. Therapeutic potential of α-glucosidase inhibitors in type 2 diabetes mellitus: an evidence-based review. Expert Opin Pharmacother. 2015;16(13):1959–81.

    Article  CAS  PubMed  Google Scholar 

  29. Narita T, Yokoyama H, Yamashita R, et al. Comparisons of the effects of 12-week administration of miglitol and voglibose on the responses of plasma incretins after a mixed meal in Japanese type 2 diabetic patients. Diabetes Obes Metab. 2012;14(3):283–7.

    Article  CAS  PubMed  Google Scholar 

  30. Miyake K, Takashi Y, Matsuzawa Y, et al. A case of severe postprandial hypoglycemia suspected to be caused by hepatitis B and liver cirrhosis. J Jpn Diabetes Soc. 2014;57(9):714–21.

    Google Scholar 

  31. Meneilly GS, Cheung E, Tuokko H. Altered responses to hypoglycemia of healthy elderly people. J Clin Endocrinol Metab. 1994;78(6):1341–8.

    CAS  PubMed  Google Scholar 

  32. Shnayder MM, Dervishi M, Jo A, et al. Congenital portosystemic shunt occlusion with an Amplatzer PFO occlusion device: a case report. CVIR Endovasc. 2021;4(1):14.

    Article  PubMed  PubMed Central  Google Scholar 

  33. Imhof A, Schneemann M, Schaffner A, et al. Reactive hypoglycaemia due to late dumping syndrome: successful treatment with acarbose. Swiss Med Wkly. 2001;131(5–6):81–3.

    CAS  PubMed  Google Scholar 

  34. Kihara Y, Ogami Y, Tabaru A, et al. Safe and effective treatment of diabetes mellitus associated with chronic liver diseases with an alpha-glucosidase inhibitor, acarbose. J Gastroenterol. 1997;32(6):777–82.

    Article  CAS  PubMed  Google Scholar 

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Acknowledgements

We thank Mark Cleasby, PhD, from Edanz (https://jp.edanz.com/ac) for editing a draft of this manuscript.

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Authors and Affiliations

  1. Department of Diabetology and Endocrinology, JCHO Tokyo Shinjuku Medical Center, 5-1, Tsukudo-cho, Shinjuku-ku, Tokyo, 162-8543, Japan

    Akiko Fujiwara, Shuhei Kanda, Keisuke Ueno & Nobuo Sekine

  2. Department of Diabetes and Metabolic Diseases, The University of Tokyo Hospital, 7-3-1, Hongo, Bunkyo-ku, Tokyo, 113-8655, Japan

    Akiko Fujiwara

  3. Department of Gastroenterology, JCHO Tokyo Shinjuku Medical Center, 5-1, Tsukudo-cho, Shinjuku-ku, Tokyo, 162-8543, Japan

    Hajime Fujie

Authors
  1. Akiko Fujiwara
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  2. Shuhei Kanda
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  4. Hajime Fujie
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  5. Nobuo Sekine
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Contributions

The first draft of the manuscript was written by AF. AF, SK, KU, and HF were in charge of treatment during hospitalization. AF and SK reviewed and edited the manuscript. NS, HF, and KU contributed to discussion. All the authors read and approved the final version of the manuscript.

Corresponding author

Correspondence to Shuhei Kanda.

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No potential conflicts of interest relevant to this article were reported.

Human and animal rights

All procedures followed were in accordance with the Ethical Standards of the responsible National Committee on Human Experimentation and with the Helsinki Declaration of 1964 and later versions. Informed consent was obtained from the patient for being included in the study.

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Fujiwara, A., Kanda, S., Ueno, K. et al. Reactive hypoglycemia owing to an intrahepatic congenital portosystemic shunt in an older patient. Diabetol Int 14, 298–303 (2023). https://doi.org/10.1007/s13340-023-00627-z

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  • DOI: https://doi.org/10.1007/s13340-023-00627-z

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