Insulinoma in Patients with Diabetes- A Systematic Review of Previously Reported Cases

Introduction: Paradoxical co-existence of insulinoma and diabetes is extremely rare. Although a few case reports addressed this association, a comprehensive study elucidating this relationship has been lacking. We performed a systematic review of published cases of insulinoma in diabetes. Methods: W...

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Main Authors: Subhankar Chatterjee, Rana Bhattacharjee, Ritwik Ghosh, Partha P. Chakraborty, Anirban Sinha, Animesh Maiti
Format: Article
Language:English
Published: Wolters Kluwer Medknow Publications 2024-12-01
Series:Indian Journal of Endocrinology and Metabolism
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Online Access:https://journals.lww.com/10.4103/ijem.ijem_154_24
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author Subhankar Chatterjee
Rana Bhattacharjee
Ritwik Ghosh
Partha P. Chakraborty
Anirban Sinha
Animesh Maiti
author_facet Subhankar Chatterjee
Rana Bhattacharjee
Ritwik Ghosh
Partha P. Chakraborty
Anirban Sinha
Animesh Maiti
author_sort Subhankar Chatterjee
collection DOAJ
description Introduction: Paradoxical co-existence of insulinoma and diabetes is extremely rare. Although a few case reports addressed this association, a comprehensive study elucidating this relationship has been lacking. We performed a systematic review of published cases of insulinoma in diabetes. Methods: We conducted a literature search using PubMed and Google Scholar, employing various combinations of the following terms: ‘insulinoma’, ‘diabetes’, ‘nesidioblastosis’, ‘endogenous hyperinsulinism’, ‘hypoglycaemia’, and ‘hyperglycaemia’ (from January 1900 to January 30, 2024). Exclusion criteria included non-English publications, duplicate articles, reports lacking sufficient data, cases of endogenous hyperinsulinemic hypoglycaemia other than insulinoma, and inaccessible articles. Statistical analysis was performed using appropriate methods. Results: Sixty patients were considered for the final analysis. Mean age was 61 ± 15 years (range: 17–96 years) with a slight female preponderance; 88.3% had type-2 diabetes with a median duration of 8 years. The median delay in diagnosis of insulinoma was 6 months. Median blood glucose varied from 30.5 mg/dL to 235 mg/dL, with a mean HbA1c of 5.6 ± 1.3% (range: 2.9%–8.2%). Critical sampling data were available in 75% of cases. The median size of the insulinoma was 2 cm. Furthermore, 5.2% of insulinomas were extra-pancreatic. Among pancreatic insulinomas, 14.5% were multi-focal. One-third of cases were malignant. Surgical resection was done in 70.9% of cases, while 40% received drug therapy and 12.7% received both, with 20.7% overall mortality. Malignant insulinoma (P = 0.007), micro-angiopathic (P = 0.018) and macro-angiopathic complications (P = 0.039), and other co-morbidities (P = 0.009) were associated with unfavourable prognosis, while being overweight and obese (P = 0.020) at presentation was associated with favourable prognosis. Conclusion: This first systematic review provides insights into the uniqueness of insulinoma in diabetes.
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spelling doaj-art-266d31a7e145465694c739690b4dba302025-01-09T12:22:14ZengWolters Kluwer Medknow PublicationsIndian Journal of Endocrinology and Metabolism2230-82102230-95002024-12-0128655456110.4103/ijem.ijem_154_24Insulinoma in Patients with Diabetes- A Systematic Review of Previously Reported CasesSubhankar ChatterjeeRana BhattacharjeeRitwik GhoshPartha P. ChakrabortyAnirban SinhaAnimesh MaitiIntroduction: Paradoxical co-existence of insulinoma and diabetes is extremely rare. Although a few case reports addressed this association, a comprehensive study elucidating this relationship has been lacking. We performed a systematic review of published cases of insulinoma in diabetes. Methods: We conducted a literature search using PubMed and Google Scholar, employing various combinations of the following terms: ‘insulinoma’, ‘diabetes’, ‘nesidioblastosis’, ‘endogenous hyperinsulinism’, ‘hypoglycaemia’, and ‘hyperglycaemia’ (from January 1900 to January 30, 2024). Exclusion criteria included non-English publications, duplicate articles, reports lacking sufficient data, cases of endogenous hyperinsulinemic hypoglycaemia other than insulinoma, and inaccessible articles. Statistical analysis was performed using appropriate methods. Results: Sixty patients were considered for the final analysis. Mean age was 61 ± 15 years (range: 17–96 years) with a slight female preponderance; 88.3% had type-2 diabetes with a median duration of 8 years. The median delay in diagnosis of insulinoma was 6 months. Median blood glucose varied from 30.5 mg/dL to 235 mg/dL, with a mean HbA1c of 5.6 ± 1.3% (range: 2.9%–8.2%). Critical sampling data were available in 75% of cases. The median size of the insulinoma was 2 cm. Furthermore, 5.2% of insulinomas were extra-pancreatic. Among pancreatic insulinomas, 14.5% were multi-focal. One-third of cases were malignant. Surgical resection was done in 70.9% of cases, while 40% received drug therapy and 12.7% received both, with 20.7% overall mortality. Malignant insulinoma (P = 0.007), micro-angiopathic (P = 0.018) and macro-angiopathic complications (P = 0.039), and other co-morbidities (P = 0.009) were associated with unfavourable prognosis, while being overweight and obese (P = 0.020) at presentation was associated with favourable prognosis. Conclusion: This first systematic review provides insights into the uniqueness of insulinoma in diabetes.https://journals.lww.com/10.4103/ijem.ijem_154_24endogenous hyperinsulinemic hypoglycaemiainsulinomanesidioblastosistype-1 diabetestype-2 diabetes
spellingShingle Subhankar Chatterjee
Rana Bhattacharjee
Ritwik Ghosh
Partha P. Chakraborty
Anirban Sinha
Animesh Maiti
Insulinoma in Patients with Diabetes- A Systematic Review of Previously Reported Cases
Indian Journal of Endocrinology and Metabolism
endogenous hyperinsulinemic hypoglycaemia
insulinoma
nesidioblastosis
type-1 diabetes
type-2 diabetes
title Insulinoma in Patients with Diabetes- A Systematic Review of Previously Reported Cases
title_full Insulinoma in Patients with Diabetes- A Systematic Review of Previously Reported Cases
title_fullStr Insulinoma in Patients with Diabetes- A Systematic Review of Previously Reported Cases
title_full_unstemmed Insulinoma in Patients with Diabetes- A Systematic Review of Previously Reported Cases
title_short Insulinoma in Patients with Diabetes- A Systematic Review of Previously Reported Cases
title_sort insulinoma in patients with diabetes a systematic review of previously reported cases
topic endogenous hyperinsulinemic hypoglycaemia
insulinoma
nesidioblastosis
type-1 diabetes
type-2 diabetes
url https://journals.lww.com/10.4103/ijem.ijem_154_24
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AT ritwikghosh insulinomainpatientswithdiabetesasystematicreviewofpreviouslyreportedcases
AT parthapchakraborty insulinomainpatientswithdiabetesasystematicreviewofpreviouslyreportedcases
AT anirbansinha insulinomainpatientswithdiabetesasystematicreviewofpreviouslyreportedcases
AT animeshmaiti insulinomainpatientswithdiabetesasystematicreviewofpreviouslyreportedcases