Clinical paradox: insulinoma in a patient with type 1 diabetes mellitus

A. S. Ametov; M. M. Tavobilov; A. A. Karpov; E. I. Zakurdaev; E. Y. Pashkova; D. M. Antsiferova

doi:10.14341/DM13240

Clinical paradox: insulinoma in a patient with type 1 diabetes mellitus

A. S. Ametov, M. M. Tavobilov, A. A. Karpov, E. I. Zakurdaev, E. Y. Pashkova, D. M. Antsiferova

https://doi.org/10.14341/DM13240

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Abstract

Insulinoma is a rare neuroendocrine tumor occurring from beta-cells of the pancreas. Тhe diagnosis of insulinoma may be difficult, especially in patients with concomitant diabetes mellitus. The reason for the similar clinical picture in these patients could be an incorrect scheme of hypoglycemic therapy. There is a number of cases of insulinoma in patients with type 2 diabetes mellitus described in literature. A more paradoxical clinical situation is the development of insulinoma in a patient with type 1 diabetes mellitus (T1DM). Analyzing a huge amount of literature sources, only three articles describing insulinoma in patients with T1DM were discovered. In all the described cases, the tumors turned out to be malignant. We present the first described in literature clinical case of the diagnosis and treatment of insulinoma in a patient with latent autoimmune diabetes in adults (LADA-diabetes), as well as the review of the literature on this topic. A gradual decrease in the need for exogenous insulin in combination with recurrent hypoglycemia made it possible to suspect the diagnosis, which was confirmed during a fasting test and instrumental examination of the tumor using computer tomography and endoscopic ultrasound.

Keywords

insulinoma, type 1 diabetes mellitus, latent autoimmune diabetes in adults, diagnosis, clinical case

About the Authors

A. S. Ametov

Russian Medical Academy of Continuing Professional Education
Russian Federation

Alexander S. Ametov - MD, PhD, Professor.

Moscow

Competing Interests:

None

M. M. Tavobilov

Botkin Hospital
Russian Federation

Mikhail M. Tavobilov - MD, PhD.

Moscow

Competing Interests:

None

A. A. Karpov

Botkin Hospital
Russian Federation

Alexey A. Karpov - MD.

Moscow

Competing Interests:

None

E. I. Zakurdaev

Botkin Hospital
Russian Federation

Evgeniy I. Zakurdaev - MD, PhD.

Moscow

Competing Interests:

None

E. Y. Pashkova

Russian Medical Academy of Continuing Professional Education; Botkin Hospital
Russian Federation

Evgeniya Y. Pashkova - MD, PhD, Associate Professor.

5 Botkinskiy proezd, 125284 Moscow

Competing Interests:

None

D. M. Antsiferova

Russian Medical Academy of Continuing Professional Education; Endocrinological Dispensary
Russian Federation

Daria M. Antsiferova - MD, PhD student.

Moscow

Competing Interests:

None

References

1. Wilder RM, Allan FN, Power MH, et al. Carcinoma of the islands of the pancreas: hyperinsulinism and hypoglycemia. JAMA. 1927;89(5):348-355. doi: https://doi.org/10.1001/JAMA.1927.02690050014007

2. Crippa S, Zerbi A, Boninsegna L, et al. Surgical management of insulinomas: short- and long-term outcomes after enucleations and pancreatic resections. Arch Surg. 2012;147(3):261-266. doi: https://doi.org/10.1001/ARCHSURG.2011.1843

3. Service FJ, McMahon MM, O’Brien Peter C, et al. Functioning insulinoma--incidence, recurrence, and long-term survival of patients: a 60-year study. Mayo Clin Proc. 1991;66(7):711-719. doi: https://doi.org/10.1016/S0025-6196(12)62083-7

4. Giannis D, Moris D, Karachaliou GS, et al. Insulinomas: from diagnosis to treatment. A review of the literature. J BUON. 2020;25(3):1302-1314

5. Rostambeigi N, Thompson GB. What should be done in an operating room when an insulinoma cannot be found? Clin Endocrinol (Oxf ). 2009;70(4):512-515. doi: https://doi.org/10.1111/J.1365-2265.2009.03527.X

6. Kane LA, Grant CS, Nippoldt TB, et al. Insulinoma in a patient with NIDDM. Diabetes Care. 1993;16(9):1298-1300. doi: https://doi.org/10.2337/DIACARE.16.9.1298

7. Svartberg J, Stridsberg M, Wilander E, et al. Tumour-induced hypoglycaemia in a patient with insulin-dependent diabetes mellitus. J Intern Med. 1996;239(2):181-185. doi: https://doi.org/10.1046/J.1365-2796.1996.405750000.X

8. Oikawa Y, Katsuki T, Kawasaki M, et al. Insulinoma may mask the existence of Type 1 diabetes. Diabet Med. 2012;29(7):e138-41. doi: https://doi.org/10.1111/J.1464-5491.2012.03615.X

9. Zwicker K, Chatten C, Gratton K, et al. Spontaneous autoimmunity sufficiently potent to induce diabetes mellitus is insufficient to protect against insulinoma. J Immunol. 2009;183(3):1705-1714. doi: https://doi.org/10.4049/JIMMUNOL.0800902

10. Lablanche S, Chobert-Bakouline M, Risse O, et al. Malignant insulinoma may arise during the course of type 1 diabetes mellitus: A case report. Diabetes Metab. 2015;41(3):258-261. doi: https://doi.org/10.1016/J.DIABET.2014.08.004

11. Roep BO, Arden SD, De Vries RRP, et al. T-cell clones from a type-1 diabetes patient respond to insulin secretory granule proteins. Nature. 1990;345(6276):632-634. doi: https://doi.org/10.1038/345632A0

12. Huang SW, MacLaren NK. Insulin-dependent diabetes: a disease of autoaggression. Science. 1976;192(4234):64-66. doi: https://doi.org/10.1126/SCIENCE.769160

13. Ishida-Oku M, Iwase M, Sugitani A, et al. A case of recurrent type 1 diabetes mellitus with insulitis of transplanted pancreas in simultaneous pancreas-kidney transplantation from cardiac death donor. Diabetologia. 2010;53(2):341-345. doi: https://doi.org/10.1007/S00125-009-1593-3

14. Vliet E Van, Roep BO, Meulenbroek L, et al. Human T cell clones with specificity for insulinoma cell antigens. Eur J Immunol. 1989;19(1):213-216. doi: https://doi.org/10.1002/EJI.1830190136

15. Monetini L, Cavallo MG, Barone F, et al. T cell reactivity to human insulinoma cell line (CM) antigens in patients with type 1 diabetes. Autoimmunity. 1999;29(3):171-177. doi: https://doi.org/10.3109/08916939908998532

16. Bashratyan R, Sheng H, Regn D, et al. Insulinoma-released exosomes activate autoreactive marginal zone-like B cells that expand endogenously in prediabetic NOD mice. Eur J Immunol. 2013;43(10):2588-2597. doi: https://doi.org/10.1002/EJI.201343376

Supplementary files

	1. Figure 1. Self-monitoring results of glycemia.
	Subject
	Type	Исследовательские инструменты
	View (359KB)	Indexing metadata ▾

	2. Figure 2. Endoscopic ultrasound (Endo-US).
	Subject
	Type	Исследовательские инструменты
	View (670KB)	Indexing metadata ▾

	3. Figure 3. Continuous glucose monitoring results before surgery.
	Subject
	Type	Исследовательские инструменты
	View (905KB)	Indexing metadata ▾

	4. Figure 4. Resected pancreatic head tumor.
	Subject
	Type	Исследовательские инструменты
	View (358KB)	Indexing metadata ▾

	5. Figure 5. Histological image of pancreatic neuroendocrine tumor. Hematoxylin and eosin staining, magnification ×5–×20.
	Subject
	Type	Исследовательские инструменты
	View (3MB)	Indexing metadata ▾

	6. Figure 6. Immunohistochemical staining of neuroendocrine tumor tissue with Synaptophysin (a–b), Chromogranin (c–d), CD56 (e–f), Ki-67 (g–h).
	Subject
	Type	Исследовательские инструменты
	View (3MB)	Indexing metadata ▾

	7. Figure 7. Continuous glucose monitoring results during basal-bolus insulin therapy after pancreatectomy.
	Subject
	Type	Исследовательские инструменты
	View (253KB)	Indexing metadata ▾

Review

For citations:

Ametov A.S., Tavobilov M.M., Karpov A.A., Zakurdaev E.I., Pashkova E.Y., Antsiferova D.M. Clinical paradox: insulinoma in a patient with type 1 diabetes mellitus. Diabetes mellitus. 2025;28(3):295-304. (In Russ.) https://doi.org/10.14341/DM13240

This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License (CC BY-NC-ND 4.0).

ISSN 2072-0351 (Print)
ISSN 2072-0378 (Online)

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	Title	Figure 1. Self-monitoring results of glycemia.
	Type	Исследовательские инструменты
	Date	2025-07-25
Digital Object Identifier	https://doi.org/10.14341/DM13240-7531

	Title	Figure 2. Endoscopic ultrasound (Endo-US).
	Type	Исследовательские инструменты
	Date	2025-07-25
Digital Object Identifier	https://doi.org/10.14341/DM13240-7532

	Title	Figure 3. Continuous glucose monitoring results before surgery.
	Type	Исследовательские инструменты
	Date	2025-07-25
Digital Object Identifier	https://doi.org/10.14341/DM13240-7533

	Title	Figure 4. Resected pancreatic head tumor.
	Type	Исследовательские инструменты
	Date	2025-07-25
Digital Object Identifier	https://doi.org/10.14341/DM13240-7534

	Title	Figure 5. Histological image of pancreatic neuroendocrine tumor. Hematoxylin and eosin staining, magnification ×5–×20.
	Type	Исследовательские инструменты
	Date	2025-07-25
Digital Object Identifier	https://doi.org/10.14341/DM13240-7535

	Title	Figure 6. Immunohistochemical staining of neuroendocrine tumor tissue with Synaptophysin (a–b), Chromogranin (c–d), CD56 (e–f), Ki-67 (g–h).
	Type	Исследовательские инструменты
	Date	2025-07-25
Digital Object Identifier	https://doi.org/10.14341/DM13240-7536

	Title	Figure 7. Continuous glucose monitoring results during basal-bolus insulin therapy after pancreatectomy.
	Type	Исследовательские инструменты
	Date	2025-07-25
Digital Object Identifier	https://doi.org/10.14341/DM13240-7537

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Diabetes mellitus

Clinical paradox: insulinoma in a patient with type 1 diabetes mellitus

Full Text:

Abstract

Keywords

About the Authors

References

Supplementary files

Review

For citations:

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