ArticleAmerican journal of clinical pathology2016
Enhanced Islet Cell Nucleomegaly Defines Diffuse Congenital Hyperinsulinism in Infancy but Not Other Forms of the Disease.
Article in American journal of clinical pathology, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 23 papers.
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Who cites it
23 citing papers in PubMed.
- Comprehensive clinical and molecular characterization with long-term outcomes in 40 patients with congenital hyperinsulinism.Endocrine · 2025Article
- Reassessing Pancreatectomy in Diffuse Congenital Hyperinsulinism: A Tale of 2 Brothers With HomozygousJCEM case reports · 2025Article
- Clinical and Genetic Characteristics of Congenital Hyperinsulinism in Norway: A Nationwide Cohort Study.The Journal of clinical endocrinology and metabolism · 2025Article
- Genotype-histotype-phenotype correlations in hyperinsulinemic hypoglycemia.Histology and histopathology · 2024Review
- Standardised practices in the networked management of congenital hyperinsulinism: a UK national collaborative consensus.Frontiers in endocrinology · 2023Review
- Non-coding variants disrupting a tissue-specific regulatory element in HK1 cause congenital hyperinsulinism.Nature genetics · 2022Article
- Variation in Glycemic Outcomes in Focal Forms of Congenital Hyperinsulinism-The UK Perspective.Journal of the Endocrine Society · 2022Article
- Congenital hyperinsulinism: localization of a focal lesion withPediatric radiology · 2022Review
- Article
- SUR1-mutant iPS cell-derived islets recapitulate the pathophysiology of congenital hyperinsulinism.Diabetologia · 2021Article
- Differential Morphological Diagnosis of Various Forms of Congenital Hyperinsulinism in Children.Frontiers in endocrinology · 2021Article
- Unravelling the genetic causes of mosaic islet morphology in congenital hyperinsulinism.The journal of pathology. Clinical research · 2020Article
- Therapies and outcomes of congenital hyperinsulinism-induced hypoglycaemia.Diabetic medicine : a journal of the British Diabetic Association · 2019Review
- Clinical Diversity in Focal Congenital Hyperinsulinism in Infancy Correlates With Histological Heterogeneity of Islet Cell Lesions.Frontiers in endocrinology · 2018Article
- Congenital Hyperinsulinism: Diagnosis and Treatment Update.Journal of clinical research in pediatric endocrinology · 2017Review
- Morphoproteomics and biomedical analytics coincide with clinical outcomes in supporting a constant but variable role for the mTOR pathway in the biology of congenital hyperinsulinism of infancy.Orphanet journal of rare diseases · 2017Article
- Atypical Forms of Congenital Hyperinsulinism in Infancy Are Associated With Mosaic Patterns of Immature Islet Cells.The Journal of clinical endocrinology and metabolism · 2017Article
- Functional and Metabolomic Consequences of KDiabetes · 2017Article
- Hyperinsulinemic hypoglycemia: clinical, molecular and therapeutical novelties.Journal of inherited metabolic disease · 2017Review
- Diagnosis and treatment of hyperinsulinaemic hypoglycaemia and its implications for paediatric endocrinology.International journal of pediatric endocrinology · 2017Review
Corrections and comments
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Authors and funding
15 authors.
Funding
Abstract
objectivesTo quantify islet cell nucleomegaly in controls and tissues obtained from patients with congenital hyperinsulinism in infancy (CHI) and to examine the association of nucleomegaly with proliferation.
methodsHigh-content analysis of histologic sections and serial block-face scanning electron microscopy were used to quantify nucleomegaly.
resultsEnlarged islet cell nuclear areas were 4.3-fold larger than unaffected nuclei, and the mean nuclear volume increased to approximately threefold. Nucleomegaly was a normal feature of pediatric islets and detected in the normal regions of the pancreas from patients with focal CHI. The incidence of nucleomegaly was highest in diffuse CHI (CHI-D), with more than 45% of islets containing two or more affected cells. While in CHI-D nucleomegaly was negatively correlated with cell proliferation, in all other cases, there was a positive correlation.
conclusionsIncreased incidence of nucleomegaly is pathognomonic for CHI-D, but these cells are nonproliferative, suggesting a novel role in the pathobiology of this condition.
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