Natural history and neurodevelopmental outcomes in perinatal stress induced hyperinsulinism

ObjectiveTo describe perinatal stress induced hyperinsulinism (PSIHI), determine the prevalence of neurodevelopmental differences, and identify risk factors for poor developmental prognosis.MethodsSubjects with a history of hyperinsulinism (HI) and perinatal stress and in whom resolution of the HI w...

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Main Authors: Winnie M. Sigal, Ohoud Alzahrani, Gabriela M. Guadalupe, Herodes Guzman, Jerilynn Radcliffe, Nina H. Thomas, Abbas F. Jawad, Diva D. De Leon
Format: Article
Language:English
Published: Frontiers Media S.A. 2022-10-01
Series:Frontiers in Pediatrics
Subjects:
Online Access:https://www.frontiersin.org/articles/10.3389/fped.2022.999274/full
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author Winnie M. Sigal
Winnie M. Sigal
Ohoud Alzahrani
Gabriela M. Guadalupe
Herodes Guzman
Jerilynn Radcliffe
Jerilynn Radcliffe
Nina H. Thomas
Nina H. Thomas
Nina H. Thomas
Abbas F. Jawad
Abbas F. Jawad
Diva D. De Leon
Diva D. De Leon
author_facet Winnie M. Sigal
Winnie M. Sigal
Ohoud Alzahrani
Gabriela M. Guadalupe
Herodes Guzman
Jerilynn Radcliffe
Jerilynn Radcliffe
Nina H. Thomas
Nina H. Thomas
Nina H. Thomas
Abbas F. Jawad
Abbas F. Jawad
Diva D. De Leon
Diva D. De Leon
author_sort Winnie M. Sigal
collection DOAJ
description ObjectiveTo describe perinatal stress induced hyperinsulinism (PSIHI), determine the prevalence of neurodevelopmental differences, and identify risk factors for poor developmental prognosis.MethodsSubjects with a history of hyperinsulinism (HI) and perinatal stress and in whom resolution of the HI was demonstrated were included. Medical record review, caregiver interview, and three validated developmental assessments were completed.ResultsOf the 107 subjects (75% male), 36% were born between 32 and 37 weeks. Median age of hypoglycemia presentation was 0 days. Median age at HI diagnosis was 12 days (IQR 13.5). Median length of time for initiation of definitive treatment was 14 days (IQR 14).Caregiver interviews were completed for 53 of 79 eligible subjects. Developmental concerns were reported by 51%. Neurodevelopmental assessments were completed by caregivers of 37 of the 53 enrolled subjects. The proportion of subjects scoring >1 SD and >2 SD away from the mean in the direction of concern on the major composite scores was significantly greater than in the general population (40.5% vs. 15.8%, P ≤ 0.0001 and 18.9% vs. 2.2%, P ≤ 0.0001, respectively).Male sex, small for gestational age status (SGA), and treatment with continuous feeds were associated with assessment scores >1 SD from the mean (P < 0.05). SGA and preeclampsia were associated with assessment scores >2 SD from the mean (P < 0.05).ConclusionWhile the majority of infants presented with hypoglycemia in the first day of life, diagnosis and treatment occurred 12–14 days later. Children with PSIHI are at high risk of neurodevelopmental deficits and are more likely to perform below average on developmental assessment.
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spelling doaj.art-3440bddcf088413ca69b314033c022782022-12-22T04:34:44ZengFrontiers Media S.A.Frontiers in Pediatrics2296-23602022-10-011010.3389/fped.2022.999274999274Natural history and neurodevelopmental outcomes in perinatal stress induced hyperinsulinismWinnie M. Sigal0Winnie M. Sigal1Ohoud Alzahrani2Gabriela M. Guadalupe3Herodes Guzman4Jerilynn Radcliffe5Jerilynn Radcliffe6Nina H. Thomas7Nina H. Thomas8Nina H. Thomas9Abbas F. Jawad10Abbas F. Jawad11Diva D. De Leon12Diva D. De Leon13Division of Endocrinology and Diabetes, Children’s Hospital of Philadelphia, Philadelphia, PA, United StatesDepartment of Pediatrics, The Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, United StatesDivision of Endocrinology and Diabetes, Children’s Hospital of Philadelphia, Philadelphia, PA, United StatesDivision of Endocrinology and Diabetes, Children’s Hospital of Philadelphia, Philadelphia, PA, United StatesDivision of Endocrinology and Diabetes, Children’s Hospital of Philadelphia, Philadelphia, PA, United StatesBehavioral Neuroscience Core, Center for Human Phenomic Science, The Children’s Hospital of Philadelphia, Philadelphia, PA, United StatesDivision of Developmental and Behavioral Pediatrics, The Children's Hospital of Philadelphia, Philadelphia, PA, United StatesBehavioral Neuroscience Core, Center for Human Phenomic Science, The Children’s Hospital of Philadelphia, Philadelphia, PA, United StatesDivision of Child and Adolescent Psychiatry, The Children's Hospital of Philadelphia, Philadelphia, PA, United StatesDepartment of Psychiatry, The Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, United StatesDepartment of Pediatrics, Children's Hospital of Philadelphia, Philadelphia, PA, United StatesBiostatistics Core, Center for Human Phenomic Science, The Children’s Hospital of Philadelphia, Philadelphia, PA, United StatesDivision of Endocrinology and Diabetes, Children’s Hospital of Philadelphia, Philadelphia, PA, United StatesDepartment of Pediatrics, The Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, United StatesObjectiveTo describe perinatal stress induced hyperinsulinism (PSIHI), determine the prevalence of neurodevelopmental differences, and identify risk factors for poor developmental prognosis.MethodsSubjects with a history of hyperinsulinism (HI) and perinatal stress and in whom resolution of the HI was demonstrated were included. Medical record review, caregiver interview, and three validated developmental assessments were completed.ResultsOf the 107 subjects (75% male), 36% were born between 32 and 37 weeks. Median age of hypoglycemia presentation was 0 days. Median age at HI diagnosis was 12 days (IQR 13.5). Median length of time for initiation of definitive treatment was 14 days (IQR 14).Caregiver interviews were completed for 53 of 79 eligible subjects. Developmental concerns were reported by 51%. Neurodevelopmental assessments were completed by caregivers of 37 of the 53 enrolled subjects. The proportion of subjects scoring >1 SD and >2 SD away from the mean in the direction of concern on the major composite scores was significantly greater than in the general population (40.5% vs. 15.8%, P ≤ 0.0001 and 18.9% vs. 2.2%, P ≤ 0.0001, respectively).Male sex, small for gestational age status (SGA), and treatment with continuous feeds were associated with assessment scores >1 SD from the mean (P < 0.05). SGA and preeclampsia were associated with assessment scores >2 SD from the mean (P < 0.05).ConclusionWhile the majority of infants presented with hypoglycemia in the first day of life, diagnosis and treatment occurred 12–14 days later. Children with PSIHI are at high risk of neurodevelopmental deficits and are more likely to perform below average on developmental assessment.https://www.frontiersin.org/articles/10.3389/fped.2022.999274/fullhypoglycemiainsulinneonatesbrain damageglucose
spellingShingle Winnie M. Sigal
Winnie M. Sigal
Ohoud Alzahrani
Gabriela M. Guadalupe
Herodes Guzman
Jerilynn Radcliffe
Jerilynn Radcliffe
Nina H. Thomas
Nina H. Thomas
Nina H. Thomas
Abbas F. Jawad
Abbas F. Jawad
Diva D. De Leon
Diva D. De Leon
Natural history and neurodevelopmental outcomes in perinatal stress induced hyperinsulinism
Frontiers in Pediatrics
hypoglycemia
insulin
neonates
brain damage
glucose
title Natural history and neurodevelopmental outcomes in perinatal stress induced hyperinsulinism
title_full Natural history and neurodevelopmental outcomes in perinatal stress induced hyperinsulinism
title_fullStr Natural history and neurodevelopmental outcomes in perinatal stress induced hyperinsulinism
title_full_unstemmed Natural history and neurodevelopmental outcomes in perinatal stress induced hyperinsulinism
title_short Natural history and neurodevelopmental outcomes in perinatal stress induced hyperinsulinism
title_sort natural history and neurodevelopmental outcomes in perinatal stress induced hyperinsulinism
topic hypoglycemia
insulin
neonates
brain damage
glucose
url https://www.frontiersin.org/articles/10.3389/fped.2022.999274/full
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