Difficult diagnosis and localization of focal nesidioblastosis: clinical implications of68Gallium-DOTA-D-Phe1-Tyr3-octreotide PET scanning
Open accessAbstract
Focal nesidioblastosis is a rare cause of endogenous hyperinsulinemic hypoglycemia in adults. Because it is difficult to localize and detect with current imaging modalities, nesidioblastosis is challenging for biliary-pancreatic surgeons. (68)Gallium-DOTA-D-Phe(1)-Tyr(3)-octreotide PET scanning and (111)indium-pentetreotide diethylene triamine pentaacetic acid octreotide scanning may be superior to conventional imaging modalities in determining the localization of nesidioblastosis. We report the successful surgical treatment of a 54-year-old woman with focal hyperplasia of the islets of Langerhans, who experienced frequent hypoglycemic symptoms and underwent various diagnostic examinations with different results.
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Not all cases with histologically proven nesidioblastosis exhibit the same clinical pattern; some patients have a positive fasting test and also suffer from fasting or exercise-induced hypoglycemia.
“However, not all cases with histologically proven nesidioblastosis exhibit the same clinical pattern. Some patients, e.g., have a positive fasting test and also suffer from fasting or exercise-induced hypoglycemia [18,28,29].”
68Ga DOTA-Exendin-4 PET/CT is especially helpful in distinguishing occult or ectopic insulinoma from NIPHS and to separate focal and diffuse nesidioblastosis.
“The latter is especially helpful in distinguishing occult or ectopic insulinoma from NIPHS and to separate focal and diffuse nesidioblastosis [29].”
From An Uncommon Cause of Recurrent Presyncope, Dizziness, and Tachycardia: A Case Report of Diffuse, Adult-Onset Nesidioblastosis/Non-Insulinoma Pancreatogenous Hypoglycemia Syndrome (NIPHS) (2023) · cites this paper as [29]
The Case Report and Discussion describe 68Ga-DOTATOC PET localizing a lesion in one patient with focal nesidioblastosis, not 68Ga DOTA-Exendin-4 PET/CT. The Discussion mentions NIPHS and the focal and diffuse forms of nesidioblastosis, but does not report that the claimed scan distinguishes insulinoma from NIPHS or separates the two forms (pages unavailable in our copy). (LLM, conf=0.76)
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Research works printed but not linked: 10
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- [2]Laidlaw GF Nesidioblastoma, the islet tumor of the pancreas Am J Pathol 1938 14 125 134 19970380 PMC1964945
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- [4]McElroy MK Lowy AM Weidner N Case report: focal nesidioblastosis ("nesidioblastoma") in an adult Hum Pathol 2010 41 447 451 20004945
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- [6]Guettier JM Kam A Chang R Skarulis MC Cochran C Alexander HR Localization of insulinomas to regions of the pancreas by intraarterial calcium stimulation: the NIH experience J Clin Endocrinol Metab 2009 94 1074 1080 19190102 PMC2682461
- [7]Doppman JL Miller DL Chang R Shawker TH Gorden P Norton JA Insulinomas: localization with selective intraarterial injection of calcium Radiology 1991 178 237 241 1984311
- [8]Srirajaskanthan R Kayani I Quigley AM Soh J Caplin ME Bomanji J The role of 68Ga-DOTATATE PET in patients with neuroendocrine tumors and negative or equivocal findings on 111In-DTPA-octreotide scintigraphy J Nucl Med 2010 51 875 882 20484441
- [9]Buchmann I Henze M Engelbrecht S Eisenhut M Runz A Schafer M Comparison of 68Ga-DOTATOC PET and 111In-DTPAOC (Octreoscan) SPECT in patients with neuroendocrine tumours Eur J Nucl Med Mol Imaging 2007 34 1617 1626 17520251
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Dieterle, M. P., Husari, A., Prozmann, S. N., Wiethoff, H., Stenzinger, A., Röhrich, M., Pfeiffer, U., Kießling, W. R., Engel, H., Sourij, H., et al. (2023) · Biomedicines · doi:10.3390/biomedicines11061741 · Cited by 5 · see citing works
“The latter is especially helpful in distinguishing occult or ectopic insulinoma from NIPHS and to separate focal and diffuse nesidioblastosis [29].”
Background
- Authors
- Kim, J. R., Jang, J.-Y., Shin, Y. C., Cho, Y. M., Kim, H., Kwon, W., Han, Y. M., & Kim, S.-W.
- Affiliations
- Seoul National University (KR)
- Corresponding author
- Jang, Jin-Young
- ORCID
- Venue
- Annals of Surgical Treatment and Research
- Publisher
- The Korean Surgical Society
- Published
- 2016
- Volume
- 91
- Issue
- 1
- Pages
- 51
- ISSN
- 2288-6575 (ISSN-L)
- 2288-6796
- PMC
- PMC4942539
- OpenAlex
- W2469143616
- Cited by
- 9
- References
- 0
- Citations by year
- 2024: 3 · 2023: 4 · 2020: 2
- FWCI
- 0.00
- Type
- journal article
- Language
- en
- Open access
- hybrid
- doi.org
- Licence
- Locations
- Annals of Surgical Treatment and Research (publishedVersion, cc-by-nc)
- Europe PMC (PubMed Central) (publishedVersion, cc-by-nc)
- PubMed Central (publishedVersion, cc-by-nc)
- PubMed (publishedVersion)
- Funders
- Norwegian Institute of Public Health
- Topics
- Health Sciences › Medicine › Epidemiology › Neuroendocrine Tumor Research Advances
- Health Sciences › Medicine › Surgery › Pancreatic function and diabetes
- Health Sciences › Medicine › Oncology › Peptidase Inhibition and Analysis
- Keywords
- endogenous hyperinsulinemic hypoglycemia, islet hyperplasia, tumor localization, pancreatic imaging, surgical treatment
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