Case Report
Copyright ©The Author(s) 2023. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Jan 6, 2023; 11(1): 150-156
Published online Jan 6, 2023. doi: 10.12998/wjcc.v11.i1.150
Adult focal β-cell nesidioblastosis: A case report
Kui Tu, Li-Jin Zhao, Jin Gu
Kui Tu, Li-Jin Zhao, Jin Gu, Department of General Surgery, Affiliated Hospital of Zunyi Medical University, Zunyi 563000, Guizhou Province, China
Author contributions: Tu K wrote the article, Zhao LJ revised the article, and Gu J reviewed the medical records and managed follow-up visits; all authors have read and approved the final version to be submitted.
Informed consent statement: Informed written consent was obtained from the patient for publication of this report and any accompanying image.
Conflict-of-interest statement: The authors declare that they have no conflict of interest.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Jin Gu, Doctor, Associate Chief Physician, Department of General Surgery, Affiliated Hospital of Zunyi Medical University, No. 149 Dalian Road, Zunyi City, Zunyi 563000, Guizhou Province, China. 64838050@qq.com
Received: September 1, 2022
Peer-review started: September 1, 2022
First decision: October 24, 2022
Revised: October 27, 2022
Accepted: December 21, 2022
Article in press: December 21, 2022
Published online: January 6, 2023
Abstract
BACKGROUND

Nesidioblastosis usually refers to a series of clinical manifestations caused by the proliferation of β-cells in pancreatic islets, and these clinical manifestations are hyperinsulinemia and persistent hypoglycemia. According to the size of the lesion, nesidioblastosis is divided into focal nesidioblastosis, diffuse nesidioblastosis and atypical nesidioblastosis, and its pathogenesis is still unclear. Nesidioblastosis is mainly seen in infants and rarely reported in adults, especially focal nesidioblastosis, which is difficult to distinguish from insulinoma.

CASE SUMMARY

We report a case of adult focal β-cell nesidioblastosis in which the preoperative diagnosis was insulinoma. The patient was a 48-year-old male who suffered from repeated morning and fasting palpitations, sweating, and severe disturbance of consciousness for 5 years. His blood glucose was found to be as low as 1.79 mmol/L during an attack. However, abdominal computed tomography showed no abnormalities. Magnetic resonance imaging and endoscopic ultrasonography demonstrated a nodular mass in the head of the pancreas, combined with hyperinsulinemia and high serum C-peptide. The patient was diagnosed with insulinoma and underwent Beger surgery; however, the postoperative pathological results showed nesidioblastosis.

CONCLUSION

Although surgical resection is the preferred option for nesidioblastosis, some cases can be treated non-surgically. In order to increase clinicians' understanding of nesidioblastosis, it is necessary to review the pathogenesis, diagnosis and treatment of this disease.

Keywords: Focal nesidioblastosis, Hyperinsulinemia, Persistent hypoglycemia, Case report

Core Tip: Focal nesidioblastosis is a neuroendocrine disease which is rarely reported in adults and easily confused with insulinoma, and its pathogenesis is still unclear. We report a rare case of adult focal β-cell nesidioblastosis, which was diagnosed as insulinoma before surgery. Increased glucose level after resection of the lesion may assist in the diagnosis of focal β-cell nesidioblastosis, due to intra-pancreatic regulation in focal nesidioblastosis. We also review the related literature. This case report may enhance clinicians' understanding of this disease.