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      Nesidioblastosis: an uncommon complication seen post Roux-en-Y gastric bypass

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          Abstract

          Summary

          Adult-onset nesidioblastosis is a rare complication of Roux-en-Y gastric bypass surgery and may occur months to years after the initial surgical procedure. It is manifested by a hyperinsulinemic, hypoglycemic state. The annual incidence of adult-onset hyperinsulinemic hypoglycemia is believed to be less than 0.1 in 1 000 000 with a mean age of onset of 47 years ( 1). Here, we describe a patient who presented with worsening hypoglycemic symptoms for 1 year prior to presentation that eventually progressed to hypoglycemic seizures. The onset of this hypoglycemia was 5 years after Roux-en-Y gastric bypass surgery. A full neurological evaluation, which included an EEG, head CT, and MRI, was performed to rule out epilepsy and other seizure-related disorders. After hypoglycemia was confirmed, extensive laboratory studies were obtained to elucidate the cause of the hypoglycemia and differentiate nesidioblastosis from insulinoma. Once the diagnosis of nesidioblastosis was established, a sub-total pancreatectomy was performed, and the patient was discharged and placed on acarbose, a competitive reversible inhibitor of pancreatic α-amylase and intestinal brush border α-glucosidases which slows carbohydrate absorption. The lack of information and understanding of nesidioblastosis due to its rarity makes any knowledge of this rare but important surgical complication essential. As incidence of obesity increases, the number of gastric bypasses being performed increases with it, and understanding this disease process will be essential for the primary care provider. This is the primary reason for the writing of this publication.

          Learning points
          • Nesidioblastosis is a persistent hyperinsulinemic, hypoglycemic state, mostly seen after Roux-en-Y gastric bypass surgery, with symptoms occurring postprandially.

          • The incidence is 0.1–0.3% of all post Roux-en-Y gastric bypass patients.

          • The key diagnostic clue to identifying nesidioblastosis is a positive selective arterial calcium stimulation test, showing a diffuse pattern of increased basal hepatic venous insulin concentration, whereas insulinomas would show focal increases.

          • Pathological specimen of pancreas will show diffuse hypertrophy of beta cells.

          • Management includes acarbose and total or subtotal pancreatectomy, which can be curative.

          • With the prevalence of obesity increasing and more patients turning to Roux-en-Y gastric bypass, more patients may be at risk of this potential surgical complication.

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          Most cited references10

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          Hypoglycemia after Gastric Bypass Surgery: Current Concepts and Controversies

          Context Hypoglycemia, occurring after bariatric and other forms of upper gastrointestinal surgery, is increasingly encountered by clinical endocrinologists. The true frequency of this condition remains uncertain, due, in part, to differences in the diagnostic criteria and in the affected populations, as well as relative lack of patient and physician awareness and understanding of this condition. Postbariatric hypoglycemia can be severe and disabling for some patients, with neuroglycopenia (altered cognition, seizures, and loss of consciousness) leading to falls, motor vehicle accidents, and job and income loss. Moreover, repeated episodes of hypoglycemia can result in hypoglycemia unawareness, further impairing safety and requiring the assistance of others to treat hypoglycemia. Objective In this review, we summarize and integrate data from studies of patients affected by hypoglycemia after Roux-en-Y gastric bypass (RYGB) surgery, obtained from PubMed searches (1990 to 2017) and reference searches of relevant retrieved articles. Whereas hypoglycemia can also be observed after sleeve gastrectomy and fundoplication, this review is focused on post-RYGB, given the greater body of published clinical studies at present. Outcome Measures Data addressing specific aspects of diagnosis, pathophysiology, and treatment were reviewed by the authors; when not available, the authors have provided opinions based on clinical experience with this challenging condition. Conclusions Hypoglycemia, occurring after gastric bypass surgery, is challenging for patients and physicians alike. This review provides a systematic approach to diagnosis and treatment based on the underlying pathophysiology. Hypoglycemia is increasingly recognized after gastric bypass. This review summarizes the pathophysiology of this challenging syndrome and provides suggested approaches to diagnosis and treatment.
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            Hyperinsulinemic hypoglycemia with nesidioblastosis after gastric-bypass surgery.

            We describe six patients (five women and one man; median age, 47 years; range, 39 to 54) with postprandial symptoms of neuroglycopenia owing to endogenous hyperinsulinemic hypoglycemia after Roux-en-Y gastric bypass surgery. Except for equivocal evidence in one patient, there was no radiologic evidence of insulinoma. Selective arterial calcium-stimulation tests, positive in each patient, were used to guide partial pancreatectomy. Nesidioblastosis was identified in resected specimens from each patient, and multiple insulinomas were identified in one. Hypoglycemic symptoms diminished postoperatively. We speculate that hyperfunction of pancreatic islets did not lead to obesity but that beta-cell trophic factors may have increased as a result of gastric bypass.
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              ASMBS Position Statement on Postprandial Hyperinsulinemic Hypoglycemia after Bariatric Surgery.

                Author and article information

                Journal
                Endocrinol Diabetes Metab Case Rep
                Endocrinol Diabetes Metab Case Rep
                EDM
                Endocrinology, Diabetes & Metabolism Case Reports
                Bioscientifica Ltd (Bristol )
                2052-0573
                05 December 2022
                2022
                : 2022
                : 22-0361
                Affiliations
                [1 ]VCOM-Auburn , 910 S Donahue Dr, Auburn, AL
                [2 ]FACP , Grandview Medical Center, 3570 Grandview Pkwy #100a, Birmingham, AL
                [3 ]FACP , Princeton Baptist Medical Center, 701 Princeton Ave SW, Birmingham, AL
                Author notes
                Correspondence should be addressed to J L Greenspan; Email: jgreenspan@ 123456auburn.vcom.edu
                Author information
                http://orcid.org/0000-0003-4309-3094
                Article
                EDM220361
                10.1530/EDM-22-0361
                9875027
                36571473
                603e1938-a5cf-41a2-8f3b-1082aaf10b90
                © The authors

                This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License..

                History
                : 19 September 2022
                : 05 December 2022
                Categories
                Adult
                Female
                White
                United States
                Pancreas
                Stomach
                Duodenum
                Diabetes
                Obesity
                New Disease or Syndrome: Presentations/Diagnosis/Management
                New Disease or Syndrome: Presentations/Diagnosis/Management

                adult,female,white,united states,pancreas,stomach,duodenum,diabetes,obesity,new disease or syndrome: presentations/diagnosis/management,december,2022

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