Insulin autoimmune syndrome misdiagnosed as an insulinoma in a woman presenting with a pancreatic cystic lesion and taki
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LETTER TO THE EDITOR
Insulin autoimmune syndrome misdiagnosed as an insulinoma in a woman presenting with a pancreatic cystic lesion and taking alpha lipoic acid: a lesson to be learned Damiano Gullo 1,2 & Miriam Magliozzo 1 & Adriana Strano 1 & Vincenzo Gaetano Piazza 3 & Gaspare Stabile 4 & Antonino Belfiore 1 Received: 9 October 2020 / Accepted: 6 November 2020 # Hellenic Endocrine Society 2020
Dear Editor, Insulinoma is a functioning pancreatic neuroendocrine tumor (PNET) originating from islet cells of the pancreas and presenting mostly as a benign and solid lesion, rarely as a cystic lesion [1]. Insulin autoimmune syndrome (IAS) is an uncommon cause of hypoglycemia among non-Asian populations, although it is also increasingly reported in Western countries associated with the use of alpha lipoic acid (ALA) [2]. In March 2020, a 71-year-old Caucasian female was referred to the Surgical Unit of the Garibaldi-Nesima Hospital, Catania, Italy, with the indication to undergo surgery for a cystic insulinoma causing hypoglycemic crises. Twenty-five days earlier, the patient had, in fact, been admitted to the emergency department of another hospital, her symptoms being diaphoresis, tremors, palpitations, and occasional loss of consciousness over the past month, partially resolved by increasing food intake. At admission, blood laboratory tests showed a low glucose level of 39 mg/dL (normal values 70–110 mg/dL). She had no history of diabetes or insulin treatment. After infusion of 10% dextrose, the patient was transferred to the Internal Medicine Unit. During an episode of symptomatic hypoglycemia, glucose level was 44 mg/ dl and serum insulin level was > 600 μIU/mL (normal values 2– 10 μIU/mL); C-peptide assay was not available at the time. The initial diagnosis was insulinoma, the most common cause of endogenous hyperinsulinemic hypoglycemia. The patient was * Damiano Gullo [email protected] 1
Endocrine Unit, Department of Clinical and Experimental Medicine, University of Catania, Garibaldi-Nesima Hospital, Catania, Italy
2
Endocrine Unit, Garibaldi-Nesima Hospital, Via Palermo 636, I-95131 Catania, Italy
3
Surgical Oncology Unit, Garibaldi-Nesima Hospital, Catania, Italy
4
Internal Medicine Unit, Umberto I Hospital, Enna, Italy
submitted to an abdominal computed tomography (CT) scan with contrast, which demonstrated a 9 × 18mm cystic lesion in the tail of the pancreas with septal and capsular enhancement. A presumptive diagnosis of a rare form of cystic insulinoma was made (1). The patient underwent distal pancreatectomy with the excision of the cyst shown on the CT scan (Fig. 1). During surgical laparoscopy, no other lesion was detected by ultrasonography. The intraoperative histological examination confirmed the cystic nature of the pancreatic lesion. During the days after surgery, however, recurrent hypoglycemic episodes persisted, requiring continuous administration of 10% intravenous dextrose. The patient was therefore transferred to the Endocrine Unit for further investigation. Laboratory tes
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