PMID- 35838801 OWN - NLM STAT- PubMed-not-MEDLINE LR - 20220718 IS - 2730-6011 (Electronic) IS - 2730-6011 (Linking) VI - 13 IP - 1 DP - 2022 Jul 15 TI - Complete remission of recurrent multiple insulin-producing neuroendocrine tumors of the pancreas with somatostatin analogs: a case report and literature review. PG - 66 LID - 10.1007/s12672-022-00531-z [doi] LID - 66 AB - Hyperinsulinemic hypoglycemia is most commonly caused by a single, sporadic insulinoma. Multicentric insulinoma disease (insulinomatosis) as well as metachronous neuroendocrine tumors of the pancreas, known also as neuroendocrine adenomatosis, represent a very rare condition, if not associated with multiple endocrine neoplasia type 1 syndrome (MEN1) or Von Hippel Lindau disease. We report a 9-year follow-up of a 41-year-old woman, initially presenting with hypoglycemic syndrome caused by two insulin-producing tumors, who underwent subtotal pancreasectomy in 2012, with histology compatible with multiple small neuroendocrine tumors. An approximately 1-cm insulin-producing tumor recurred at subsequent biochemical and radiological follow-up, and was cured with the somatostatin analog octreotide as a single treatment, until remission of symptoms and complete regression of the pancreatic lesion achieved after only 16 months of treatment. The possible mechanisms for these findings are discussed and the literature is briefly reviewed. CI - (c) 2022. The Author(s). FAU - Tartaglia, Andreas AU - Tartaglia A AD - Unit of Endocrinology and Metabolism, Morgagni Hospital, Via Carlo Forlanini 34, 47100, Forli, Italy. a.tartaglia@ausl.fo.it. FAU - Busonero, Giulia AU - Busonero G AD - Unit of Endocrinology and Metabolism, Morgagni Hospital, Via Carlo Forlanini 34, 47100, Forli, Italy. FAU - Gagliardi, Lorenza AU - Gagliardi L AD - Unit of Endocrinology and Metabolism, Morgagni Hospital, Via Carlo Forlanini 34, 47100, Forli, Italy. FAU - Boddi, Valentina AU - Boddi V AD - Unit of Endocrinology and Metabolism, Morgagni Hospital, Via Carlo Forlanini 34, 47100, Forli, Italy. FAU - Pieri, Federica AU - Pieri F AD - Unit of Pathology, Morgagni Hospital, Forli, Italy. FAU - Nizzoli, Maurizio AU - Nizzoli M AD - Unit of Endocrinology and Metabolism, Morgagni Hospital, Via Carlo Forlanini 34, 47100, Forli, Italy. LA - eng PT - Journal Article DEP - 20220715 PL - United States TA - Discov Oncol JT - Discover oncology JID - 101775142 PMC - PMC9287506 OTO - NOTNLM OT - Hypoglycemia OT - Multicentric insulinomas OT - Pancreatic neuroendocrine tumors OT - Somatostatin analogs COIS- The authors declare no potential competing interests. EDAT- 2022/07/16 06:00 MHDA- 2022/07/16 06:01 PMCR- 2022/07/15 CRDT- 2022/07/15 11:55 PHST- 2022/05/18 00:00 [received] PHST- 2022/07/06 00:00 [accepted] PHST- 2022/07/15 11:55 [entrez] PHST- 2022/07/16 06:00 [pubmed] PHST- 2022/07/16 06:01 [medline] PHST- 2022/07/15 00:00 [pmc-release] AID - 10.1007/s12672-022-00531-z [pii] AID - 531 [pii] AID - 10.1007/s12672-022-00531-z [doi] PST - epublish SO - Discov Oncol. 2022 Jul 15;13(1):66. doi: 10.1007/s12672-022-00531-z.