Diagnosis of pancreatic focal nesidioblastosis assisted by dual‑nuclide tracer positron emission tomography/computed tomography: A case report

Zhu,Guangwen; Xie,Liting; Hu,Xianwen

doi:10.3892/etm.2023.12330

Diagnosis of pancreatic focal nesidioblastosis assisted by dual‑nuclide tracer positron emission tomography/computed tomography: A case report

Authors:
- Guangwen Zhu
- Liting Xie
- Xianwen Hu
View Affiliations

Affiliations: Department of Endocrinology, Zunyi Hospital of Traditional Chinese Medicine, Zunyi, Guizhou 563000, P.R. China, Department of Gynaecology, Zunyi Hospital of Traditional Chinese Medicine, Zunyi, Guizhou 563000, P.R. China, Department of Nuclear Medicine, The Affiliated Hospital of Zunyi Medical University, Zunyi, Guizhou 563003, P.R. China
Published online on: November 28, 2023 https://doi.org/10.3892/etm.2023.12330
Article Number: 42
Copyright: © Zhu et al. This is an open access article distributed under the terms of Creative Commons Attribution License.

Metrics: Total Views: 0 (Spandidos Publications: | PMC Statistics: )

Metrics: Total PDF Downloads: 0 (Spandidos Publications: | PMC Statistics: )

Cited By (CrossRef): 0 citations Loading Articles...

Abstract

Nesidioblastosis is a rare cause of hyperinsulinemic hypoglycemia in adults and its clinical features are similar to those of insulinoma with recurrent hypoglycemic attacks. The present study reports the case of a 48‑year‑old man who visited the Affiliated Hospital of Zunyi Medical University (Zunyi, China) with a 5‑year history of recurrent hypoglycemic symptoms such as dizziness and palpitations. Abdominal magnetic resonance imaging (MRI) showed a mass of ~1.2x1.0 cm in the head of the pancreas, which was suspected to be an insulinoma. For confirmation, the patient underwent both fluorine‑18‑fluorodeoxyglucose (¹⁸F‑FDG) and gallium‑68‑labeled 1,4,7,10‑tetraazacyclododecane‑1,4,7,10‑teraacetic acid‑d‑Phel‑Tyr3‑Thr8‑OC (⁶⁸Ga‑DOTATATE) positron emission tomography/computed tomography (PET/CT), which showed a moderately increased uptake of ¹⁸F‑FDG but no uptake of ⁶⁸Ga‑DOTATATE in the corresponding lesion. The patient subsequently underwent surgery to remove the lesion, which was pathologically confirmed as a pancreatic nesidioblastosis. This case showed that nesidioblastosis should be considered a differential diagnosis for insulinoma and that dual nuclear tracer PET/CT imaging is helpful for differentiating between the two. If conventional imaging techniques such as ultrasound, CT and MRI cannot identify the cause of hypoglycemia in future cases, dual‑nuclide tracer PET/CT imaging should be considered.

View Figures

View References

1	Laidlaw GF: Nesidioblastoma, the islet tumor of the pancreas. Am J Pathol. 14:125–134.5. 1938.PubMed/NCBI
2	García-Santos EP, Manzanares-Campillo Mdel C, Padilla-Valverde D, Villarejo-Campos P, Gil-Rendo A, Muñoz-Atienza V, Sánchez-García S, Puig-Rullán AM, Rodríguez-Peralto JL and Martín-Fernández J: Nesidioblastosis. A case of hyperplasia of the islets of Langerhans in the adult. Pancreatology. 13:544–548. 2013.PubMed/NCBI View Article : Google Scholar
3	Klöppel G, Anlauf M, Raffel A, Perren A and Knoefel WT: Adult diffuse nesidioblastosis: Genetically or environmentally induced? Hum Pathol. 39:3–8. 2008.PubMed/NCBI View Article : Google Scholar
4	Yamada Y, Kitayama K, Oyachi M, Higuchi S, Kawakita R, Kanamori Y and Yorifuji T: Nationwide survey of endogenous hyperinsulinemic hypoglycemia in Japan (2017-2018): Congenital hyperinsulinism, insulinoma, non-insulinoma pancreatogenous hypoglycemia syndrome and insulin autoimmune syndrome (Hirata's disease). J Diabetes Investig. 11:554–563. 2020.PubMed/NCBI View Article : Google Scholar
5	Anlauf M, Wieben D, Perren A, Sipos B, Komminoth P, Raffel A, Kruse ML, Fottner C, Knoefel WT, Mönig H, et al: Persistent hyperinsulinemic hypoglycemia in 15 adults with diffuse nesidioblastosis: Diagnostic criteria, incidence, and characterization of beta-cell changes. Am J Surg Pathol. 29:524–533. 2005.PubMed/NCBI View Article : Google Scholar
6	Witteles RM, Straus II FH, Sugg SL, Koka MR, Costa EA and Kaplan EL: Adult-onset nesidioblastosis causing hypoglycemia: An important clinical entity and continuing treatment dilemma. Arch Surg. 136:656–663. 2001.PubMed/NCBI View Article : Google Scholar
7	Prasad V, Sainz-Esteban A, Arsenic R, Plöckinger U, Denecke T, Pape UF, Pascher A, Kühnen P, Pavel M and Blankenstein O: Role of (68)Ga somatostatin receptor PET/CT in the detection of endogenous hyperinsulinaemic focus: An explorative study. Eur J Nucl Med Mol Imaging. 43:1593–1600. 2016.PubMed/NCBI View Article : Google Scholar
8	Dravecka I and Lazurova I: Nesidioblastosis in adults. Neoplasma. 61:252–256. 2014.PubMed/NCBI View Article : Google Scholar
9	Glaser B, Chiu KC, Anker R, Nestorowicz A, Landau H, Ben-Bassat H, Shlomai Z, Kaiser N, Thornton PS, Stanley CA, et al: Familial hyperinsulinism maps to chromosome 11p14-15.1, 30 cM centromeric to the insulin gene. Nat Genet. 7:185–188. 1994.PubMed/NCBI View Article : Google Scholar
10	Rumilla KM, Erickson LA, Service FJ, Vella A, Thompson GB, Grant CS and Lloyd RV: Hyperinsulinemic hypoglycemia with nesidioblastosis: Histologic features and growth factor expression. Mod Pathol. 22:239–245. 2009.PubMed/NCBI View Article : Google Scholar
11	Dieterle MP, Husari A, Prozmann SN, Wiethoff H, Stenzinger A, Röhrich M, Pfeiffer U, Kießling WR, Engel H, Sourij H, et al: Diffuse, adult-onset nesidioblastosis/non-insulinoma pancreatogenous hypoglycemia syndrome (NIPHS): Review of the literature of a rare cause of hyperinsulinemic hypoglycemia. Biomedicines. 11(1732)2023.PubMed/NCBI View Article : Google Scholar
12	Ahmed FW, Majeed MS and Kirresh O: Non-Diabetic Hypoglycemia. In: StatPearls [Internet]. Treasure Island (FL): StatPearls Publishing; 2023.
13	Geraghty M, Draman M, Moran D, Muldoon C, Reynolds JV and Cullen MJ: Hypoglycaemia in an adult male: A surprising finding in pursuit of insulinoma. Surgeon. 6:57–60. 2008.PubMed/NCBI View Article : Google Scholar
14	Starke A, Saddig C, Kirch B, Tschahargane C and Goretzki P: Islet hyperplasia in adults: Challenge to preoperatively diagnose non-insulinoma pancreatogenic hypoglycemia syndrome. World J Surg. 30:670–679. 2006.PubMed/NCBI View Article : Google Scholar
15	Doi S, Yamada T, Kito Y, Obara S, Fujii Y, Nishimura T, Kato T, Nakayama H, Tsutsumi M and Okamura R: Adult-Onset Focal Nesidioblastosis With Nodular Formation Mimicking Insulinoma. J Endocr Soc. 6(bvab185)2021.PubMed/NCBI View Article : Google Scholar
16	Mehrabi A, Fischer L, Hafezi M, Dirlewanger A, Grenacher L, Diener MK, Fonouni H, Golriz M, Garoussi C, Fard N, et al: A systematic review of localization, surgical treatment options, and outcome of insulinoma. Pancreas. 43:675–686. 2014.PubMed/NCBI View Article : Google Scholar
17	Hu X, Li D, Wang R, Wang P and Cai J: Comparison of the application of 18F-FDG and 68Ga-DOTATATE PET/CT in neuroendocrine tumors: A retrospective study. Medicine (Baltimore). 102(e33726)2023.PubMed/NCBI View Article : Google Scholar
18	Deroose CM, Hindié E, Kebebew E, Goichot B, Pacak K, Taïeb D and Imperiale A: Molecular imaging of gastroenteropancreatic neuroendocrine tumors: Current status and future directions. J Nucl Med. 57:1949–1956. 2016.PubMed/NCBI View Article : Google Scholar
19	Kalff V, Iravani A, Akhurst T, Pattison DA, Eu P, Hofman MS and Hicks RJ: Utility of ⁶⁸Ga-DOTA-Exendin-4 positron emission tomography-computed tomography imaging in distinguishing between insulinoma and nesidioblastosis in patients with confirmed endogenous hyperinsulinaemic hypoglycaemia. Intern Med J. 51:1657–1664. 2021.PubMed/NCBI View Article : Google Scholar
20	Antwi K, Hepprich M, Müller NA, Reubi JC, Fani M, Rottenburger C, Nicolas G, Kaul F, Christ ER and Wild D: Pitfalls in the detection of insulinomas with glucagon-like peptide-1 receptor imaging. Clin Nucl Med. 45:e386–e392. 2020.PubMed/NCBI View Article : Google Scholar
21	Dauriz M, Maneschi C, Castelli C, Tomezzoli A, Fuini A, Landoni L, Malleo G, Ferdeghini M, Bonora E and Moghetti P: A case report of insulinoma relapse on background nesidioblastosis: A rare cause of adult hypoglycemia. J Clin Endocrinol Metab. 104:773–778. 2019.PubMed/NCBI View Article : Google Scholar
22	Arao T, Okada Y, Hirose A and Tanaka Y: A rare case of adult-onset nesidioblastosis treated successfully with diazoxide. Endocr J. 53:95–100. 2006.PubMed/NCBI View Article : Google Scholar
23	Demartin S, Goffette P, Christ E, Freitag MT, Maiter D and Maria Furnica R: Adult-onset nesidioblastosis: A challenging diagnosis revealed by glucagon-like-peptide-1 receptor imaging. Endocrinol Diabetes Metab Case Rep. 2022:22–0325. 2022.PubMed/NCBI View Article : Google Scholar
24	Nagtegaal ID, Odze RD, Klimstra D, Paradis V, Rugge M, Schirmacher P, Washington KM, Carneiro F and Cree IA: WHO Classification of Tumours Editorial Board. The 2019 WHO classification of tumours of the digestive system. Histopathology. 76:182–188. 2020.PubMed/NCBI View Article : Google Scholar