The effect of hypoglycaemia on neurocognitive outcome in children and adolescents with transient or persistent congenital hyperinsulinism
Aim To examine the hypoglycaemic effect on neurodevelopmental outcome in patients with transient and persistent congenital hyperinsulinism (CHI) born in the 21st century. Method A cohort of 117 patients (66 males, 51 females) with CHI aged 5 to 16 years (mean age 8y 11mo, SD 2y 7mo) were selected fr...
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| Veröffentlicht in: | Developmental medicine and child neurology 2019-04, Vol.61 (4), p.451-457 |
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| creator | Muukkonen, Liisa Männistö, Jonna Jääskeläinen, Jarmo Hannonen, Riitta Huopio, Hanna |
| description | Aim
To examine the hypoglycaemic effect on neurodevelopmental outcome in patients with transient and persistent congenital hyperinsulinism (CHI) born in the 21st century.
Method
A cohort of 117 patients (66 males, 51 females) with CHI aged 5 to 16 years (mean age 8y 11mo, SD 2y 7mo) were selected from a Finnish nationwide registry to examine all the patients with similar methods. Neurodevelopment was first evaluated retrospectively. The 83 patients with no risk factors for neurological impairment other than hypoglycaemia were recruited and 44 participated (24 males, 20 females; mean age 9y 7mo, SD 3y 1mo) in neuropsychological assessment with the Wechsler Intelligence Scale for Children, Fourth Edition and the Finnish version of the Developmental Neuropsychological Assessment, Second Edition domains of attention, language, memory, sensorimotor, and visual functioning.
Results
In retrospective analysis, transient and persistent CHI groups had similar prevalences of mild (22% and 18% respectively) or severe (5% and 7% respectively) neurodevelopmental difficulties. In clinical assessment, the neurocognitive profile was within the average range in both groups, but children with persistent CHI showed significant but restricted deficits in attention, memory, visual, and sensorimotor functions compared with the general population. The transient CHI group did not differ from the standardization samples.
Interpretation
Besides the more apparent broader neurological deficits, children with persistent CHI have an increased risk for milder specific neurocognitive problems, which should be considered in the follow‐up.
What this paper adds
Children with persistent congenital hyperinsulinism showed deficits in attention, memory, visual, and sensorimotor functions.
The deficits were potentially of hypoglycaemic origin.
Children with transient hyperinsulinism did not differ from the general population.
Resumen
El efecto de la hipoglucemia sobre el resultado neurocognitivo en niños y adolescentes con hiperinsulinismo congénito transitorio o persistente
Objetivo
Examinar el efecto hipoglucémico sobre el resultado del neurodesarrollo en pacientes nacidos en el siglo XXI con hiperinsulinismo congénito (HIC) transitorio y persistente.
Método
Una cohorte de 117 pacientes (66 varones, 51 mujeres) con HIC de 5 a 16 años de edad (media de 8 años 11 meses, DS 2 años 7 meses) fueron seleccionados de un registro nacional finlandés para examinar a todos los pacientes con métodos simila |
| doi_str_mv | 10.1111/dmcn.14039 |
| format | Article |
| fullrecord | <record><control><sourceid>proquest_cross</sourceid><recordid>TN_cdi_proquest_miscellaneous_2111745570</recordid><sourceformat>XML</sourceformat><sourcesystem>PC</sourcesystem><sourcerecordid>2111745570</sourcerecordid><originalsourceid>FETCH-LOGICAL-c3659-938e78d9ca229d16c8b05b3cbeb9fec19b7fc602f9e4e3c5aca65b7109fc04503</originalsourceid><addsrcrecordid>eNp9kMtO4zAUQK0Ro2mnsOEDkJdopHTsxHl4iToPRiqwKevIublpjRy72AmonzB_PQ7tsMQb60pHx9eHkEvOljye720PdskFy-QnMueikElVCnlG5ozxNOFFms7I1xCeGGNZkYsvZJaxVBQiq-bk72aHFLsOYaCuo7vD3m3NART2WlFnqcXRO3Bbqwf9gtSNA7geqbYUdtq0Hi1VtqWqdQYDoB0CfdXDjg5e2aDjTJ2ne_RBh2GawNktRpky01votQ2j0VaH_px87pQJeHG6F-Tx18_N6jZZP_z-s7pZJxCXl4nMKiyrVoJKU9nyAqqG5U0GDTYy_oLLpuygYGknUWAGuQJV5E3JmeyAiZxlC3J99O69ex4xDHWv4-bGKItuDHUak5Yiz8sJ_XZEwbsQPHb13ute-UPNWT2lr6f09Vv6CF-dvGPTY_uO_m8dAX4EXrXBwweq-sfd6v4o_QdbY5Me</addsrcrecordid><sourcetype>Aggregation Database</sourcetype><iscdi>true</iscdi><recordtype>article</recordtype><pqid>2111745570</pqid></control><display><type>article</type><title>The effect of hypoglycaemia on neurocognitive outcome in children and adolescents with transient or persistent congenital hyperinsulinism</title><source>MEDLINE</source><source>Wiley Online Library Journals Frontfile Complete</source><source>Wiley Online Library Free Content</source><creator>Muukkonen, Liisa ; Männistö, Jonna ; Jääskeläinen, Jarmo ; Hannonen, Riitta ; Huopio, Hanna</creator><creatorcontrib>Muukkonen, Liisa ; Männistö, Jonna ; Jääskeläinen, Jarmo ; Hannonen, Riitta ; Huopio, Hanna</creatorcontrib><description>Aim
To examine the hypoglycaemic effect on neurodevelopmental outcome in patients with transient and persistent congenital hyperinsulinism (CHI) born in the 21st century.
Method
A cohort of 117 patients (66 males, 51 females) with CHI aged 5 to 16 years (mean age 8y 11mo, SD 2y 7mo) were selected from a Finnish nationwide registry to examine all the patients with similar methods. Neurodevelopment was first evaluated retrospectively. The 83 patients with no risk factors for neurological impairment other than hypoglycaemia were recruited and 44 participated (24 males, 20 females; mean age 9y 7mo, SD 3y 1mo) in neuropsychological assessment with the Wechsler Intelligence Scale for Children, Fourth Edition and the Finnish version of the Developmental Neuropsychological Assessment, Second Edition domains of attention, language, memory, sensorimotor, and visual functioning.
Results
In retrospective analysis, transient and persistent CHI groups had similar prevalences of mild (22% and 18% respectively) or severe (5% and 7% respectively) neurodevelopmental difficulties. In clinical assessment, the neurocognitive profile was within the average range in both groups, but children with persistent CHI showed significant but restricted deficits in attention, memory, visual, and sensorimotor functions compared with the general population. The transient CHI group did not differ from the standardization samples.
Interpretation
Besides the more apparent broader neurological deficits, children with persistent CHI have an increased risk for milder specific neurocognitive problems, which should be considered in the follow‐up.
What this paper adds
Children with persistent congenital hyperinsulinism showed deficits in attention, memory, visual, and sensorimotor functions.
The deficits were potentially of hypoglycaemic origin.
Children with transient hyperinsulinism did not differ from the general population.
Resumen
El efecto de la hipoglucemia sobre el resultado neurocognitivo en niños y adolescentes con hiperinsulinismo congénito transitorio o persistente
Objetivo
Examinar el efecto hipoglucémico sobre el resultado del neurodesarrollo en pacientes nacidos en el siglo XXI con hiperinsulinismo congénito (HIC) transitorio y persistente.
Método
Una cohorte de 117 pacientes (66 varones, 51 mujeres) con HIC de 5 a 16 años de edad (media de 8 años 11 meses, DS 2 años 7 meses) fueron seleccionados de un registro nacional finlandés para examinar a todos los pacientes con métodos similares. El neurodesarrollo se evaluó por primera vez de forma retrospectiva. Los 83 pacientes sin factores de riesgo para el deterioro neurológico distintos de la hipoglucemia fueron reclutados y 44 de ellos participaron (24 varones, 20 mujeres; edad media 9 años 7 meses, DS 3 años 1mes) en la evaluación neuropsicológica con la Escala de Inteligencia de Wechsler y la versión finlandesa de la Evaluación Neuropsicológica del desarrollo, segunda edición, incluyendo los dominios de atención, lenguaje, memoria, sensoriomotor y funcionamiento visual.
Resultados
En el análisis retrospectivo, los grupos de HIC transitorios y persistentes tuvieron prevalencias similares de dificultades del neurodesarrollo leves (22% y 18% respectivamente) o graves (5% y 7% respectivamente). En la evaluación clínica, el perfil neurocognitivo estuvo dentro del rango promedio en ambos grupos, pero los niños con HIC persistente mostraron déficits significativos pero restringidos en la atención, memoria, funciones visuales y sensomotrices en comparación con la población general. El grupo de HIC transitorio no difirió de las muestras de estandarización.
Interpretación
Además de los déficits neurológicos más aparentes generalizados, los niños con HIC persistente tienen un mayor riesgo de presentar problemas neurocognitivos específicos más leves, que deben ser considerados en el seguimiento.
Resumo
O efeito da hipoglicemia no resultado neurocognitivo em crianças e adolescentes com hiperinsulinemia congênita transitória ou persistente
Objetivo
Examinar o efeito hipoglicêmico no resultado neurodesenvolvimental em pacientes com hyperinsulinemia congênita (HIC) transitória ou persistente nascidas no século 21.
Método
Uma coorte de 117 pacientes (66 do sexo masculino, 51 do sexo feminino) com HIC e idades de 5 a 16 anos (média de idade 8a 11m, DP 2a 7m) foram selecionados de um registro nacional finlandês para exame com métodos similares. Primeiramente, o neurodesenvolvimento foi avaliado retrospectivamente. Os 83 pacientes sem risco para deficiência neurológica além da hipoglicemia foram recrutados, e 44 participaram (24 do sexo masculino, 20 do sexo feminino; média de idade 9a 7m, DP 3a 1m) na avaliação neuropsicológica com a escala Wechsler de Inteligência, e a versão finlandesa da Avaliação Neuropsicológica Desenvolvimental, segunda edição, nos domínios de atenção, linguagem, memória, sensório‐motor, e funcionamento visual.
Resultados
Na análise retrospective, os grupos com HIC transitória e persistente tiveram prevalências similares de dificuldades neurodesenvolvimentais leves (22% e 18% respectivamente) ou severa (5% e 7% respectivamente). Na avaliação clínica, o perfil neurocognitivo estava dentro da média para ambos os grupos, mas crianças com HIC persistente mostraram deficits significantes, mas restritos, nas funções de atenção, de memória, visuais e sensório‐motoras comparado com a população em geral. O grupo com HIC transitória não diferiu das amostras padronizadas.
Interpretação
Além dos deficits neurológicos mais amplos e aparentes, crianças com HIC persistente têm risco aumentado de problemas neurocognitivos específicos leves, o que deve ser considerado no acompanhamento.
What this paper adds
Children with persistent congenital hyperinsulinism showed deficits in attention, memory, visual, and sensorimotor functions.
The deficits were potentially of hypoglycaemic origin.
Children with transient hyperinsulinism did not differ from the general population.
This article's has been translated into Spanish and Portuguese.
Follow the links from the to view the translations.</description><identifier>ISSN: 0012-1622</identifier><identifier>EISSN: 1469-8749</identifier><identifier>DOI: 10.1111/dmcn.14039</identifier><identifier>PMID: 30246438</identifier><language>eng</language><publisher>England</publisher><subject>Adolescent ; Child ; Child, Preschool ; Cognition Disorders - diagnosis ; Cognition Disorders - epidemiology ; Congenital Hyperinsulinism - psychology ; Female ; Humans ; Hypoglycemia - psychology ; Male ; Neurodevelopmental Disorders - diagnosis ; Neurodevelopmental Disorders - epidemiology ; Neuropsychological Tests ; Prevalence ; Retrospective Studies ; Wechsler Scales</subject><ispartof>Developmental medicine and child neurology, 2019-04, Vol.61 (4), p.451-457</ispartof><rights>2018 Mac Keith Press</rights><rights>2018 Mac Keith Press.</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c3659-938e78d9ca229d16c8b05b3cbeb9fec19b7fc602f9e4e3c5aca65b7109fc04503</citedby><cites>FETCH-LOGICAL-c3659-938e78d9ca229d16c8b05b3cbeb9fec19b7fc602f9e4e3c5aca65b7109fc04503</cites><orcidid>0000-0002-0596-0719</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://onlinelibrary.wiley.com/doi/pdf/10.1111%2Fdmcn.14039$$EPDF$$P50$$Gwiley$$H</linktopdf><linktohtml>$$Uhttps://onlinelibrary.wiley.com/doi/full/10.1111%2Fdmcn.14039$$EHTML$$P50$$Gwiley$$H</linktohtml><link.rule.ids>314,776,780,1411,1427,27901,27902,45550,45551,46384,46808</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/30246438$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Muukkonen, Liisa</creatorcontrib><creatorcontrib>Männistö, Jonna</creatorcontrib><creatorcontrib>Jääskeläinen, Jarmo</creatorcontrib><creatorcontrib>Hannonen, Riitta</creatorcontrib><creatorcontrib>Huopio, Hanna</creatorcontrib><title>The effect of hypoglycaemia on neurocognitive outcome in children and adolescents with transient or persistent congenital hyperinsulinism</title><title>Developmental medicine and child neurology</title><addtitle>Dev Med Child Neurol</addtitle><description>Aim
To examine the hypoglycaemic effect on neurodevelopmental outcome in patients with transient and persistent congenital hyperinsulinism (CHI) born in the 21st century.
Method
A cohort of 117 patients (66 males, 51 females) with CHI aged 5 to 16 years (mean age 8y 11mo, SD 2y 7mo) were selected from a Finnish nationwide registry to examine all the patients with similar methods. Neurodevelopment was first evaluated retrospectively. The 83 patients with no risk factors for neurological impairment other than hypoglycaemia were recruited and 44 participated (24 males, 20 females; mean age 9y 7mo, SD 3y 1mo) in neuropsychological assessment with the Wechsler Intelligence Scale for Children, Fourth Edition and the Finnish version of the Developmental Neuropsychological Assessment, Second Edition domains of attention, language, memory, sensorimotor, and visual functioning.
Results
In retrospective analysis, transient and persistent CHI groups had similar prevalences of mild (22% and 18% respectively) or severe (5% and 7% respectively) neurodevelopmental difficulties. In clinical assessment, the neurocognitive profile was within the average range in both groups, but children with persistent CHI showed significant but restricted deficits in attention, memory, visual, and sensorimotor functions compared with the general population. The transient CHI group did not differ from the standardization samples.
Interpretation
Besides the more apparent broader neurological deficits, children with persistent CHI have an increased risk for milder specific neurocognitive problems, which should be considered in the follow‐up.
What this paper adds
Children with persistent congenital hyperinsulinism showed deficits in attention, memory, visual, and sensorimotor functions.
The deficits were potentially of hypoglycaemic origin.
Children with transient hyperinsulinism did not differ from the general population.
Resumen
El efecto de la hipoglucemia sobre el resultado neurocognitivo en niños y adolescentes con hiperinsulinismo congénito transitorio o persistente
Objetivo
Examinar el efecto hipoglucémico sobre el resultado del neurodesarrollo en pacientes nacidos en el siglo XXI con hiperinsulinismo congénito (HIC) transitorio y persistente.
Método
Una cohorte de 117 pacientes (66 varones, 51 mujeres) con HIC de 5 a 16 años de edad (media de 8 años 11 meses, DS 2 años 7 meses) fueron seleccionados de un registro nacional finlandés para examinar a todos los pacientes con métodos similares. El neurodesarrollo se evaluó por primera vez de forma retrospectiva. Los 83 pacientes sin factores de riesgo para el deterioro neurológico distintos de la hipoglucemia fueron reclutados y 44 de ellos participaron (24 varones, 20 mujeres; edad media 9 años 7 meses, DS 3 años 1mes) en la evaluación neuropsicológica con la Escala de Inteligencia de Wechsler y la versión finlandesa de la Evaluación Neuropsicológica del desarrollo, segunda edición, incluyendo los dominios de atención, lenguaje, memoria, sensoriomotor y funcionamiento visual.
Resultados
En el análisis retrospectivo, los grupos de HIC transitorios y persistentes tuvieron prevalencias similares de dificultades del neurodesarrollo leves (22% y 18% respectivamente) o graves (5% y 7% respectivamente). En la evaluación clínica, el perfil neurocognitivo estuvo dentro del rango promedio en ambos grupos, pero los niños con HIC persistente mostraron déficits significativos pero restringidos en la atención, memoria, funciones visuales y sensomotrices en comparación con la población general. El grupo de HIC transitorio no difirió de las muestras de estandarización.
Interpretación
Además de los déficits neurológicos más aparentes generalizados, los niños con HIC persistente tienen un mayor riesgo de presentar problemas neurocognitivos específicos más leves, que deben ser considerados en el seguimiento.
Resumo
O efeito da hipoglicemia no resultado neurocognitivo em crianças e adolescentes com hiperinsulinemia congênita transitória ou persistente
Objetivo
Examinar o efeito hipoglicêmico no resultado neurodesenvolvimental em pacientes com hyperinsulinemia congênita (HIC) transitória ou persistente nascidas no século 21.
Método
Uma coorte de 117 pacientes (66 do sexo masculino, 51 do sexo feminino) com HIC e idades de 5 a 16 anos (média de idade 8a 11m, DP 2a 7m) foram selecionados de um registro nacional finlandês para exame com métodos similares. Primeiramente, o neurodesenvolvimento foi avaliado retrospectivamente. Os 83 pacientes sem risco para deficiência neurológica além da hipoglicemia foram recrutados, e 44 participaram (24 do sexo masculino, 20 do sexo feminino; média de idade 9a 7m, DP 3a 1m) na avaliação neuropsicológica com a escala Wechsler de Inteligência, e a versão finlandesa da Avaliação Neuropsicológica Desenvolvimental, segunda edição, nos domínios de atenção, linguagem, memória, sensório‐motor, e funcionamento visual.
Resultados
Na análise retrospective, os grupos com HIC transitória e persistente tiveram prevalências similares de dificuldades neurodesenvolvimentais leves (22% e 18% respectivamente) ou severa (5% e 7% respectivamente). Na avaliação clínica, o perfil neurocognitivo estava dentro da média para ambos os grupos, mas crianças com HIC persistente mostraram deficits significantes, mas restritos, nas funções de atenção, de memória, visuais e sensório‐motoras comparado com a população em geral. O grupo com HIC transitória não diferiu das amostras padronizadas.
Interpretação
Além dos deficits neurológicos mais amplos e aparentes, crianças com HIC persistente têm risco aumentado de problemas neurocognitivos específicos leves, o que deve ser considerado no acompanhamento.
What this paper adds
Children with persistent congenital hyperinsulinism showed deficits in attention, memory, visual, and sensorimotor functions.
The deficits were potentially of hypoglycaemic origin.
Children with transient hyperinsulinism did not differ from the general population.
This article's has been translated into Spanish and Portuguese.
Follow the links from the to view the translations.</description><subject>Adolescent</subject><subject>Child</subject><subject>Child, Preschool</subject><subject>Cognition Disorders - diagnosis</subject><subject>Cognition Disorders - epidemiology</subject><subject>Congenital Hyperinsulinism - psychology</subject><subject>Female</subject><subject>Humans</subject><subject>Hypoglycemia - psychology</subject><subject>Male</subject><subject>Neurodevelopmental Disorders - diagnosis</subject><subject>Neurodevelopmental Disorders - epidemiology</subject><subject>Neuropsychological Tests</subject><subject>Prevalence</subject><subject>Retrospective Studies</subject><subject>Wechsler Scales</subject><issn>0012-1622</issn><issn>1469-8749</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2019</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNp9kMtO4zAUQK0Ro2mnsOEDkJdopHTsxHl4iToPRiqwKevIublpjRy72AmonzB_PQ7tsMQb60pHx9eHkEvOljye720PdskFy-QnMueikElVCnlG5ozxNOFFms7I1xCeGGNZkYsvZJaxVBQiq-bk72aHFLsOYaCuo7vD3m3NART2WlFnqcXRO3Bbqwf9gtSNA7geqbYUdtq0Hi1VtqWqdQYDoB0CfdXDjg5e2aDjTJ2ne_RBh2GawNktRpky01votQ2j0VaH_px87pQJeHG6F-Tx18_N6jZZP_z-s7pZJxCXl4nMKiyrVoJKU9nyAqqG5U0GDTYy_oLLpuygYGknUWAGuQJV5E3JmeyAiZxlC3J99O69ex4xDHWv4-bGKItuDHUak5Yiz8sJ_XZEwbsQPHb13ute-UPNWT2lr6f09Vv6CF-dvGPTY_uO_m8dAX4EXrXBwweq-sfd6v4o_QdbY5Me</recordid><startdate>201904</startdate><enddate>201904</enddate><creator>Muukkonen, Liisa</creator><creator>Männistö, Jonna</creator><creator>Jääskeläinen, Jarmo</creator><creator>Hannonen, Riitta</creator><creator>Huopio, Hanna</creator><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7X8</scope><orcidid>https://orcid.org/0000-0002-0596-0719</orcidid></search><sort><creationdate>201904</creationdate><title>The effect of hypoglycaemia on neurocognitive outcome in children and adolescents with transient or persistent congenital hyperinsulinism</title><author>Muukkonen, Liisa ; Männistö, Jonna ; Jääskeläinen, Jarmo ; Hannonen, Riitta ; Huopio, Hanna</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c3659-938e78d9ca229d16c8b05b3cbeb9fec19b7fc602f9e4e3c5aca65b7109fc04503</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2019</creationdate><topic>Adolescent</topic><topic>Child</topic><topic>Child, Preschool</topic><topic>Cognition Disorders - diagnosis</topic><topic>Cognition Disorders - epidemiology</topic><topic>Congenital Hyperinsulinism - psychology</topic><topic>Female</topic><topic>Humans</topic><topic>Hypoglycemia - psychology</topic><topic>Male</topic><topic>Neurodevelopmental Disorders - diagnosis</topic><topic>Neurodevelopmental Disorders - epidemiology</topic><topic>Neuropsychological Tests</topic><topic>Prevalence</topic><topic>Retrospective Studies</topic><topic>Wechsler Scales</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Muukkonen, Liisa</creatorcontrib><creatorcontrib>Männistö, Jonna</creatorcontrib><creatorcontrib>Jääskeläinen, Jarmo</creatorcontrib><creatorcontrib>Hannonen, Riitta</creatorcontrib><creatorcontrib>Huopio, Hanna</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>MEDLINE - Academic</collection><jtitle>Developmental medicine and child neurology</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Muukkonen, Liisa</au><au>Männistö, Jonna</au><au>Jääskeläinen, Jarmo</au><au>Hannonen, Riitta</au><au>Huopio, Hanna</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>The effect of hypoglycaemia on neurocognitive outcome in children and adolescents with transient or persistent congenital hyperinsulinism</atitle><jtitle>Developmental medicine and child neurology</jtitle><addtitle>Dev Med Child Neurol</addtitle><date>2019-04</date><risdate>2019</risdate><volume>61</volume><issue>4</issue><spage>451</spage><epage>457</epage><pages>451-457</pages><issn>0012-1622</issn><eissn>1469-8749</eissn><abstract>Aim
To examine the hypoglycaemic effect on neurodevelopmental outcome in patients with transient and persistent congenital hyperinsulinism (CHI) born in the 21st century.
Method
A cohort of 117 patients (66 males, 51 females) with CHI aged 5 to 16 years (mean age 8y 11mo, SD 2y 7mo) were selected from a Finnish nationwide registry to examine all the patients with similar methods. Neurodevelopment was first evaluated retrospectively. The 83 patients with no risk factors for neurological impairment other than hypoglycaemia were recruited and 44 participated (24 males, 20 females; mean age 9y 7mo, SD 3y 1mo) in neuropsychological assessment with the Wechsler Intelligence Scale for Children, Fourth Edition and the Finnish version of the Developmental Neuropsychological Assessment, Second Edition domains of attention, language, memory, sensorimotor, and visual functioning.
Results
In retrospective analysis, transient and persistent CHI groups had similar prevalences of mild (22% and 18% respectively) or severe (5% and 7% respectively) neurodevelopmental difficulties. In clinical assessment, the neurocognitive profile was within the average range in both groups, but children with persistent CHI showed significant but restricted deficits in attention, memory, visual, and sensorimotor functions compared with the general population. The transient CHI group did not differ from the standardization samples.
Interpretation
Besides the more apparent broader neurological deficits, children with persistent CHI have an increased risk for milder specific neurocognitive problems, which should be considered in the follow‐up.
What this paper adds
Children with persistent congenital hyperinsulinism showed deficits in attention, memory, visual, and sensorimotor functions.
The deficits were potentially of hypoglycaemic origin.
Children with transient hyperinsulinism did not differ from the general population.
Resumen
El efecto de la hipoglucemia sobre el resultado neurocognitivo en niños y adolescentes con hiperinsulinismo congénito transitorio o persistente
Objetivo
Examinar el efecto hipoglucémico sobre el resultado del neurodesarrollo en pacientes nacidos en el siglo XXI con hiperinsulinismo congénito (HIC) transitorio y persistente.
Método
Una cohorte de 117 pacientes (66 varones, 51 mujeres) con HIC de 5 a 16 años de edad (media de 8 años 11 meses, DS 2 años 7 meses) fueron seleccionados de un registro nacional finlandés para examinar a todos los pacientes con métodos similares. El neurodesarrollo se evaluó por primera vez de forma retrospectiva. Los 83 pacientes sin factores de riesgo para el deterioro neurológico distintos de la hipoglucemia fueron reclutados y 44 de ellos participaron (24 varones, 20 mujeres; edad media 9 años 7 meses, DS 3 años 1mes) en la evaluación neuropsicológica con la Escala de Inteligencia de Wechsler y la versión finlandesa de la Evaluación Neuropsicológica del desarrollo, segunda edición, incluyendo los dominios de atención, lenguaje, memoria, sensoriomotor y funcionamiento visual.
Resultados
En el análisis retrospectivo, los grupos de HIC transitorios y persistentes tuvieron prevalencias similares de dificultades del neurodesarrollo leves (22% y 18% respectivamente) o graves (5% y 7% respectivamente). En la evaluación clínica, el perfil neurocognitivo estuvo dentro del rango promedio en ambos grupos, pero los niños con HIC persistente mostraron déficits significativos pero restringidos en la atención, memoria, funciones visuales y sensomotrices en comparación con la población general. El grupo de HIC transitorio no difirió de las muestras de estandarización.
Interpretación
Además de los déficits neurológicos más aparentes generalizados, los niños con HIC persistente tienen un mayor riesgo de presentar problemas neurocognitivos específicos más leves, que deben ser considerados en el seguimiento.
Resumo
O efeito da hipoglicemia no resultado neurocognitivo em crianças e adolescentes com hiperinsulinemia congênita transitória ou persistente
Objetivo
Examinar o efeito hipoglicêmico no resultado neurodesenvolvimental em pacientes com hyperinsulinemia congênita (HIC) transitória ou persistente nascidas no século 21.
Método
Uma coorte de 117 pacientes (66 do sexo masculino, 51 do sexo feminino) com HIC e idades de 5 a 16 anos (média de idade 8a 11m, DP 2a 7m) foram selecionados de um registro nacional finlandês para exame com métodos similares. Primeiramente, o neurodesenvolvimento foi avaliado retrospectivamente. Os 83 pacientes sem risco para deficiência neurológica além da hipoglicemia foram recrutados, e 44 participaram (24 do sexo masculino, 20 do sexo feminino; média de idade 9a 7m, DP 3a 1m) na avaliação neuropsicológica com a escala Wechsler de Inteligência, e a versão finlandesa da Avaliação Neuropsicológica Desenvolvimental, segunda edição, nos domínios de atenção, linguagem, memória, sensório‐motor, e funcionamento visual.
Resultados
Na análise retrospective, os grupos com HIC transitória e persistente tiveram prevalências similares de dificuldades neurodesenvolvimentais leves (22% e 18% respectivamente) ou severa (5% e 7% respectivamente). Na avaliação clínica, o perfil neurocognitivo estava dentro da média para ambos os grupos, mas crianças com HIC persistente mostraram deficits significantes, mas restritos, nas funções de atenção, de memória, visuais e sensório‐motoras comparado com a população em geral. O grupo com HIC transitória não diferiu das amostras padronizadas.
Interpretação
Além dos deficits neurológicos mais amplos e aparentes, crianças com HIC persistente têm risco aumentado de problemas neurocognitivos específicos leves, o que deve ser considerado no acompanhamento.
What this paper adds
Children with persistent congenital hyperinsulinism showed deficits in attention, memory, visual, and sensorimotor functions.
The deficits were potentially of hypoglycaemic origin.
Children with transient hyperinsulinism did not differ from the general population.
This article's has been translated into Spanish and Portuguese.
Follow the links from the to view the translations.</abstract><cop>England</cop><pmid>30246438</pmid><doi>10.1111/dmcn.14039</doi><tpages>7</tpages><orcidid>https://orcid.org/0000-0002-0596-0719</orcidid><oa>free_for_read</oa></addata></record> |
| fulltext | fulltext |
| identifier | ISSN: 0012-1622 |
| ispartof | Developmental medicine and child neurology, 2019-04, Vol.61 (4), p.451-457 |
| issn | 0012-1622 1469-8749 |
| language | eng |
| recordid | cdi_proquest_miscellaneous_2111745570 |
| source | MEDLINE; Wiley Online Library Journals Frontfile Complete; Wiley Online Library Free Content |
| subjects | Adolescent Child Child, Preschool Cognition Disorders - diagnosis Cognition Disorders - epidemiology Congenital Hyperinsulinism - psychology Female Humans Hypoglycemia - psychology Male Neurodevelopmental Disorders - diagnosis Neurodevelopmental Disorders - epidemiology Neuropsychological Tests Prevalence Retrospective Studies Wechsler Scales |
| title | The effect of hypoglycaemia on neurocognitive outcome in children and adolescents with transient or persistent congenital hyperinsulinism |
| url | https://sfx.bib-bvb.de/sfx_tum?ctx_ver=Z39.88-2004&ctx_enc=info:ofi/enc:UTF-8&ctx_tim=2025-02-07T21%3A48%3A33IST&url_ver=Z39.88-2004&url_ctx_fmt=infofi/fmt:kev:mtx:ctx&rfr_id=info:sid/primo.exlibrisgroup.com:primo3-Article-proquest_cross&rft_val_fmt=info:ofi/fmt:kev:mtx:journal&rft.genre=article&rft.atitle=The%20effect%20of%20hypoglycaemia%20on%20neurocognitive%20outcome%20in%20children%20and%20adolescents%20with%20transient%20or%20persistent%20congenital%20hyperinsulinism&rft.jtitle=Developmental%20medicine%20and%20child%20neurology&rft.au=Muukkonen,%20Liisa&rft.date=2019-04&rft.volume=61&rft.issue=4&rft.spage=451&rft.epage=457&rft.pages=451-457&rft.issn=0012-1622&rft.eissn=1469-8749&rft_id=info:doi/10.1111/dmcn.14039&rft_dat=%3Cproquest_cross%3E2111745570%3C/proquest_cross%3E%3Curl%3E%3C/url%3E&disable_directlink=true&sfx.directlink=off&sfx.report_link=0&rft_id=info:oai/&rft_pqid=2111745570&rft_id=info:pmid/30246438&rfr_iscdi=true |