ArticleOrphanet journal of rare diseases2020
Longitudinal Auxological recovery in a cohort of children with Hyperinsulinaemic Hypoglycaemia.
Article in Orphanet journal of rare diseases, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 6 papers.
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Who cites it
6 citing papers in PubMed, 12 citations in OpenAlex.
- Congenital Hyperinsulinaemic Hypoglycaemia-A Review and Case Presentation.Journal of clinical medicine · 2022Review
- Congenital hyperinsulinism in infancy and childhood: challenges, unmet needs and the perspective of patients and families.Orphanet journal of rare diseases · 2022Review
- Families' Experiences of Continuous Glucose Monitoring in the Management of Congenital Hyperinsulinism: A Thematic Analysis.Frontiers in endocrinology · 2022Article
- Clustering of Hypoglycemia Events in Patients With Hyperinsulinism: Extension of the Digital Phenotype Through Retrospective Data Analysis.Journal of medical Internet research · 2021Article
- Long-Term Outcome and Treatment in Persistent and Transient Congenital Hyperinsulinism: A Finnish Population-Based Study.The Journal of clinical endocrinology and metabolism · 2021Article
- HYPO-CHEAT's aggregated weekly visualisations of risk reduce real world hypoglycaemia.Digital healthArticle
Corrections and comments
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Authors and funding
13 authors at 5 institutions in 3 countries.
Funding
Abstract
backgroundHypoglycaemia due to hyperinsulinism (HI) is the commonest cause of severe, recurrent hypoglycaemia in childhood. Cohort outcomes of HI remain to be described and whilst previous follow up studies have focused on neurodevelopmental outcomes, there is no information available on feeding and auxology.
aimWe aimed to describe HI outcomes for auxology, medications, feeding and neurodevelopmental in a cohort up to age 5 years.
methodWe reviewed medical records for all patients with confirmed HI over a three-year period in a single centre to derive a longitudinal dataset.
resultsSeventy patients were recruited to the study. Mean weight at birth was - 1.0 standard deviation scores (SDS) for age and sex, while mean height at 3 months was - 1.5 SDS. Both weight and height trended to the population median over the follow up period. Feeding difficulties were noted in 17% of patients at 3 months and this reduced to 3% by 5 years. At age 5 years, 11 patients (15%) had neurodevelopmental delay and of these only one was severe. Resolution of disease was predicted by lower maximum early diazoxide dose (p = 0.007) and being born SGA (p = 0.009).
conclusionIn a three-year cohort of HI patients followed up for 5 years, in spite of feeding difficulties and carbohydrate loading in early life, auxology parameters are normal in follow up. A lower than expected rate of neurodevelopmental delay could be attributed to prompt early treatment.
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