ArticleJournal of diabetes investigation2020
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).
Article in Journal of diabetes investigation, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 41 papers, 1 of them a synthesis that pooled it.
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Who cites it
41 citing papers in PubMed, 1 synthesis or guideline pooled it, 65 citations in OpenAlex.
- International Guidelines for the Diagnosis and Management of Hyperinsulinism.Hormone research in paediatrics · 2024Guideline
- Alpha-lipoic acid as a trigger of insulin autoimmune syndrome: Current evidence, a case-based approach, diagnostic pitfalls, and practical management.Metabolism open · 2026Review
- Diagnostic efficacy of fasting insulin-to-C-peptide ratio in exogenous insulin autoimmune syndrome.Frontiers in medicine · 2026Article
- Hyperinsulinemic hypoglycemia due to pathogenic INSR variants: metabolic signature, phenotypic overlap, and semidominant inheritance.Frontiers in endocrinology · 2026Article
- Clinical pediatric endocrinology : case reports and clinical investigations : official journal of the Japanese Society for Pediatric Endocrinology · 2025Article
- Hirata Syndrome in a Woman With Graves' Disease Without Exposure to Thionamides and Summary of Cases from South America.JCEM case reports · 2025Article
- Insulinoma Associated with MEN1 Syndrome: A Case of Persistent Hypoglycemia in a School-aged ChildJournal of clinical research in pediatric endocrinology · 2025Article
- Neonatal hyperinsulinism: a retrospective study of presentation and management in a tertiary neonatal intensive care unit in the UK.Archives of disease in childhood. Fetal and neonatal edition · 2025Article
- Insulin autoimmune syndrome in a 3-year-old boy: the youngest to date in China and a narrow review.BMC pediatrics · 2025Review
- Insulin Autoimmune Syndrome: A Chinese Expert Consensus Statement.Aging medicine (Milton (N.S.W)) · 2025Article
- Clinically Adult-Onset Nesidioblastosis with Repeated Severe Hypoglycemia, Successfully Treated by Two Times Pancreatectomies. A Rare Case Report.Clinical and experimental gastroenterology · 2025Article
- The Birth Prevalence of Congenital Hyperinsulinism: A Narrative Review of the Epidemiology of a Rare Disease.Hormone research in paediatrics · 2025Review
- Clinical and molecular evaluation of insulin autoimmune syndrome in a woman with Graves' disease who subsequently became pregnant: A case report.Case reports in women's health · 2024Article
- Nesidioblastosis in Pregnancy: Navigating the Diagnostic and Therapeutic Challenges of a Rare Condition.Cureus · 2024Article
- Genotype-histotype-phenotype correlations in hyperinsulinemic hypoglycemia.Histology and histopathology · 2024Review
- Insulinoma presenting with anti-insulin antibodies.Endocrinology, diabetes & metabolism case reports · 2024Article
- Article
- A case of congenital hyperinsulinism presenting with diabetes after long-term diazoxide therapy.Diabetology international · 2024Article
- A clinical case of insulin autoimmune syndrome with monoclonal gammopathy of uncertain significance; complexity in management.Oxford medical case reports · 2024Article
- Diagnosis of pancreatic focal nesidioblastosis assisted by dual‑nuclide tracer positron emission tomography/computed tomography: A case report.Experimental and therapeutic medicine · 2024Article
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Authors and funding
7 authors at 2 institutions in 1 country.
Funding
Abstract
AIMS/
introductionWe aimed to investigate the nationwide incidence, treatment details and outcomes of patients with endogenous hyperinsulinemic hypoglycemia (EHH), including those with transient/persistent congenital hyperinsulinism (CHI), insulinoma, non-insulinoma pancreatogenous hypoglycemia syndrome and insulin autoimmune syndrome (Hirata's disease) in Japan. MATERIALS AND
methodsA nationwide, questionnaire-based survey was carried out to determine the number of patients with EHH who were treated for hypoglycemia or hypoglycemia-related complications in 2017-2018. The questionnaires were sent to all hospitals in Japan with >300 beds, and with pediatric and/or adult clinics likely managing EHH patients. The secondary questionnaires were sent to obtain the patients' date of birth, sex, age at onset, treatment details and post-treatment outcomes.
resultsA total of 447 patients with CHI (197 transient CHI, 225 persistent CHI and 25, unknown histology), 205 with insulinoma (118 benign, 18 malignant and 69 unknown subtype), 111 with non-insulinoma pancreatogenous hypoglycemia syndrome (33 post-gastric surgery HH, 57 postprandial HH, 10 nesidioblastosis and 11 unknown subtype) and 22 with insulin autoimmune syndrome were identified. Novel findings included: (i) marked improvement in the prognosis of persistent CHI over the past 10 years; (ii) male dominance in the incidence of transient CHI; (iii) non-insulinoma pancreatogenous hypoglycemia syndrome emerging as the second most common form of EHH in adults; (iv) frequent association of diabetes mellitus with insulin autoimmune syndrome; and (v) frequent post-treatment residual hypoglycemia and impaired quality of life.
conclusionsThe first nationwide, all age group survey of EHH showed the current status of each type of EHH disorder and the unmet needs of the patients.
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