Cited by 156Pancreatic function and diabetesDiabetes and associated disordersDiabetes Management and Research

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Hyperinsulinemic hypoglycemia of infancy (nesidioblastosis) in clinical remission: high incidence of diabetes mellitus and persistent beta-cell dysfunction at long-term follow-up

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Leibowitz, G, Glaser, B, Higazi, A A, Salameh, M, Cerasi, E, Landau, H·1995·The Journal of Clinical Endocrinology & Metabolism

DOI 10.1210/jcem.80.2.7852494

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Abstract

In persistent hyperinsulinemic hypoglycemia of infancy (PHHI), the long term outcome of the disease is not well documented. Previous reports suggested that partial pancreatectomy in infants does not endanger future islet function. We evaluated endocrine pancreatic function in 14 PHHI patients 6.5-21 yr after diagnosis. Eight underwent early subtotal pancreatectomy, and 6 were medically treated; all were in clinical remission. Intravenous glucose tolerance and C-peptide suppression tests were performed, with multiple determinations of hormone levels. The insulin response to glucose was blunted in all pancreatectomized and in 2 conservatively treated patients. Glucose disposal was reduced in 6 pancreatectomized patients and in 2 medically treated subjects. Six of the pancreatectomized patients, including two with normal glucose disposal at first evaluation, developed overt diabetes during puberty. None in the medically treated group became diabetic; however, only 2 patients have reached puberty. C-Peptide suppression in response to hypoglycemia was inadequate in 4 of 5 pancreatectomized and 3 of 5 nonpancreatectomized patients studied. These results show that children with PHHI have impaired insulin responses to glucose and lack of suppressibility of endogenous insulin secretion several years after clinical remission. Thus, the beta-cell defect responsible for the disease in infancy is not corrected with time despite the disappearance of spontaneous hypoglycemia. Insulin secret…

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Self-citation

Authors: Leibowitz, G., Glaser, B., Higazi, A. A., Salameh, M., Cerasi, E., & Landau, H.
Affiliations: Hebrew University of Jerusalem (IL)
ORCID: Leibowitz, G
Higazi, A A
Cerasi, E
Venue: The Journal of Clinical Endocrinology & Metabolism
Publisher: The Endocrine Society
Published: 1995-02
Volume: 80
Issue: 2
Pages: 386-392
ISSN: 0021-972X (ISSN-L)
1945-7197
DOI: 10.1210/jcem.80.2.7852494
OpenAlex: W1967142752
Cited by: 156
References: 32
Citations by year: 2025: 1 · 2024: 3 · 2023: 5 · 2022: 2 · 2021: 1 · 2020: 2 · 2019: 1 · 2018: 2 · 2017: 2 · 2016: 3 · 2015: 7 · 2014: 5 · 2013: 2 · 2012: 6
FWCI: 6.90
Type: journal article
Language: en
Open access: hybrid
doi.org
Licence: cc-by
Locations: The Journal of Clinical Endocrinology & Metabolism (publishedVersion, cc-by)
The Journal of Clinical Endocrinology & Metabolism (publishedVersion)
PubMed (publishedVersion)
Funders: Hadassah Medical Organization
Topics: Health Sciences › Medicine › Endocrinology, Diabetes and Metabolism › Pancreatic function and diabetes
Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Diabetes and associated disorders
Health Sciences › Medicine › Endocrinology, Diabetes and Metabolism › Diabetes Management and Research
Keywords: nesidioblastosis, diabetes mellitus, beta-cell dysfunction, subtotal pancreatectomy, glucose tolerance, glucose disposal, insulin secretion, long-term follow-up, puberty, medical treatment
MeSH terms: Adolescent, C-Peptide, Child, Diabetes Mellitus, Diazoxide, Female, Follow-Up Studies, Glucose Tolerance Test, Humans, Islets of Langerhans, Male, Pancreatectomy, Pancreatic Diseases, Prospective Studies, Time Factors, Octreotide

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Hyperinsulinemic hypoglycemia of infancy (nesidioblastosis) in clinical remission: high incidence of diabetes mellitus and persistent beta-cell dysfunction at long-term follow-up

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