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Concomitant AIMAH and multiple endocrine neoplasia type1 (MEN1) is rare to our knowledge.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Case presentation<\/jats:title>\n                <jats:p>Herein, we describe a 32\u00a0year old woman with long history of prolactinoma and secondary ammonhrea presented with not-severe manifestation of hypoglycemia due to concomitant presence of insulinoma with AIMAH leading to 12\u00a0years delay of MEN1 diagnosis. Laboratory tests showed severe hypoglycemia associated with hyper insulinemia (non-fasting blood sugar\u2009=\u200943\u00a0mg\/dl, insulin\u2009=\u200980.6 \u03bcIU \/ml, C-peptide\u2009=\u20099.3\u00a0ng\/ml) hyperparathyroidism (calcium\u2009=\u200910.3\u00a0mg\/dl, phosphor\u2009=\u20093.1\u00a0mg\/dl, PTH\u2009=\u2009280\u00a0pg\/ml) and chemical evidence of an ACTH-independent hypercortisolism (serum cortisol value of 3.5, after 1\u00a0mg dexamethasone suppression test serum ACTH value of 17\u00a0pg\/ml, and high urinary cortisol level). Abdominal CT scan demonstrated two enhancing well-defined masses 27*20\u00a0mm and 37*30\u00a0mm in the tail and body of the pancreas, respectively, and a 36*15\u00a0mm mass in left adrenal gland (seven Hounsfield units). Dynamic pituitary MRI revealed a partial empty sella. The physical examination of the patient was unremarkable. Distal pancreatectomy and a left adrenalectomy were performed. After the surgery, we observed clinical and biochemical remission of hyper insulinemia and gradual decrease in urinary cortisol. The histological features of the removed left adrenal gland were consistent with AIMAH. Histological examination of the pancreatic lesions revealed well differentiated neuroendocrine tumors. Genetic abnormalities in the <jats:italic>MEN1,<\/jats:italic> heterozygote for pathogenic variant chr11; 645,773,330-64577333AGAC, c.249-252delGTCT, p. (11e85Serfs Ter33) in exon 2 were found. It was recommended the patient undergoes parathyroidectomy as soon as possible.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Conclusion<\/jats:title>\n                <jats:p>Given the history and presentation of our case, we recommend that the clinicians consider the possibility of autonomous cortisol production in MEN1 patients who do not show severe symptoms of hypoglycemia in the presence of insulinoma.\n<\/jats:p>\n              <\/jats:sec>","DOI":"10.1186\/s12902-022-01022-6","type":"journal-article","created":{"date-parts":[[2022,4,21]],"date-time":"2022-04-21T09:06:04Z","timestamp":1650531964000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":7,"title":["Long delay in diagnosis of a case with MEN1 due to concomitant presence of AIMAH with insulinoma: a case report and literature review"],"prefix":"10.1186","volume":"22","author":[{"given":"Vajihe","family":"Chavoshi","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Seyed Saeed","family":"Tamehri Zadeh","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shayesteh","family":"Khalili","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Amirhassan","family":"Rabbani","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Seyed Amir Hassan","family":"Matini","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zhaleh","family":"Mohsenifar","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8935-2744","authenticated-orcid":false,"given":"Farzad","family":"Hadaegh","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2022,4,21]]},"reference":[{"key":"1022_CR1","doi-asserted-by":"crossref","unstructured":"Lacroix A, Bourdeau I. 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