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Ectopic insulinoma: a systematic review

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Abstract

Knowledge of ectopic insulinomas comes from single cases. We performed a systematic review through PubMed, Web of Science, Embase, eLibrary and ScienceDirect of all cases reported in the last four decades. We also describe one unreported patient. From 28 patients with ectopic insulinoma, 78.6% were female and mean age was 55.7 ± 19.2 years. Hypoglycaemia was the first symptom in 85.7% while 14.3% complained of abdominal pain or genital symptoms. Median tumour diameter was 27.5 [15-52.5] mm and it was localised by CT (73.1%), MRI (88.9%), [68Ga]Ga-DOTA-exedin-4 PET/CT (100%), 68Ga-labelled-DOTA-conjugated somatostatin analogue PET/TC (100%), somatostatin receptor scintigraphy (40%) and endoscopic ultrasound (50%). Ectopic insulinomas were located at duodenum (n = 3), jejunum (n = 2), and one respectively at stomach, liver, appendix, rectum, mesentery, ligament of Treitz, gastrosplenic ligament, hepatoduodenal ligament and splenic hilum. Seven insulinomas were affecting the female reproductive organs: ovary (n = 5), cervix (n = 2) and remaining tumours were at retroperitoneum (n = 3), kidney (n = 2), spleen (n = 1) and pelvis (n = 1). 89.3% underwent surgery (66.7% surgery vs. 33.3% laparoscopy) and 16% underwent an ineffective pancreatectomy. 85.7% had localized disease at diagnosis and 14.3% developed distant metastasis. Median follow-up time was 14.5 [4.5–35.5] months and mortality was reported in 28.6% with median time until death of 60 [5-144] months. In conclusion, ectopic insulinomas are presented as hypoglycaemia with female preponderance. Functional imaging [68Ga]Ga-DOTA-exedin-4 PET/CT and 68Ga-labelled-DOTA-conjugated somatostatin analogue PET/TC have very high sensitivity. Clinicians should be alert to the possibility of extra-pancreatic insulinomas when classic diagnostic tests and intraoperative pancreas exploration failed to locate the tumour.

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Data Availability

The data generated and analyzed during the current study are available from the corresponding author on reasonable request.

Abbreviations

CT:

Computed tomography

EUS:

Endoscopic ultrasound

GLP-1:

Glucagon-like peptide 1

IGF-2:

Insulin-like growth factor 2

MEN-1:

Multiples endocrine neoplasia type 1

MRI:

Magnetic resonance imaging

NETs:

Neuroendocrine tumours

PET:

Positron emission tomography

SRS:

Somatostatin receptor scintigraphy

US:

Ultrasonography

WHO:

World Health Organization

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Acknowledgements

Georgina F. Caulfield for the language review of the paper.

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FG-P, NV, CV (conceived the study and wrote original draft); FG-P, LH, SNC, NV (acquisition and curation data); FG-P, NV, JLV-C (analysis and interpretation data); JB, LS, JLC-C, NV (critical revision of the manuscript). All authors have read and agreed to publish the final version of the manuscript.

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Correspondence to Fernando Guerrero-Pérez.

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Guerrero-Pérez, F., Vilarrasa, N., Huánuco, L.V. et al. Ectopic insulinoma: a systematic review. Rev Endocr Metab Disord 24, 1135–1146 (2023). https://doi.org/10.1007/s11154-023-09824-2

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