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Duplication 6q24: More Than Just Diabetes

Chromosome 6q24-related transient neonatal diabetes mellitus is characterized by intrauterine growth restriction and low birth weight, with neonatal hyperglycemia resolving by 18 months and an increased risk for type 2 diabetes in adulthood. Molecularly, it is caused by overexpression of the 6q24 im...

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Autores principales: Gore, Rachel H, Nikita, Maria Eleni, Newton, Paula G, Carter, Rebecca G, Reyes-Bautista, Jeanine, Greene, Carol L
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Oxford University Press 2020
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7185952/
https://www.ncbi.nlm.nih.gov/pubmed/32373772
http://dx.doi.org/10.1210/jendso/bvaa027
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author Gore, Rachel H
Nikita, Maria Eleni
Newton, Paula G
Carter, Rebecca G
Reyes-Bautista, Jeanine
Greene, Carol L
author_facet Gore, Rachel H
Nikita, Maria Eleni
Newton, Paula G
Carter, Rebecca G
Reyes-Bautista, Jeanine
Greene, Carol L
author_sort Gore, Rachel H
collection PubMed
description Chromosome 6q24-related transient neonatal diabetes mellitus is characterized by intrauterine growth restriction and low birth weight, with neonatal hyperglycemia resolving by 18 months and an increased risk for type 2 diabetes in adulthood. Molecularly, it is caused by overexpression of the 6q24 imprinted chromosomal region due to a duplication, uniparental disomy, or abnormal methylation. Conventional testing for this condition analyzes methylation patterns at the 6q24 locus but does not evaluate for the presence of other surrounding chromosomal abnormalities. We report a female with a history of neonatal hyperglycemia due to a paternally inherited duplication at chromosomal location 6q24. She subsequently presented to the pediatric genetics clinic at 15 months of age with developmental delay and abnormal balance. Microarray analysis identified a larger 14 Mb chromosomal duplication from 6q24 to 6q25.2, consistent with a diagnosis of duplication 6q syndrome. This case highlights the clinical importance of pursuing further genetic evaluation in patients diagnosed with chromosome 6q24-related neonatal hyperglycemia via targeted methylation-specific multiplex ligation-dependent probe amplification analysis identifying a duplication in this region. Early identification and intervention can improve developmental outcomes for patients with larger chromosome 6q duplications.
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spelling pubmed-71859522020-05-05 Duplication 6q24: More Than Just Diabetes Gore, Rachel H Nikita, Maria Eleni Newton, Paula G Carter, Rebecca G Reyes-Bautista, Jeanine Greene, Carol L J Endocr Soc Case Report Chromosome 6q24-related transient neonatal diabetes mellitus is characterized by intrauterine growth restriction and low birth weight, with neonatal hyperglycemia resolving by 18 months and an increased risk for type 2 diabetes in adulthood. Molecularly, it is caused by overexpression of the 6q24 imprinted chromosomal region due to a duplication, uniparental disomy, or abnormal methylation. Conventional testing for this condition analyzes methylation patterns at the 6q24 locus but does not evaluate for the presence of other surrounding chromosomal abnormalities. We report a female with a history of neonatal hyperglycemia due to a paternally inherited duplication at chromosomal location 6q24. She subsequently presented to the pediatric genetics clinic at 15 months of age with developmental delay and abnormal balance. Microarray analysis identified a larger 14 Mb chromosomal duplication from 6q24 to 6q25.2, consistent with a diagnosis of duplication 6q syndrome. This case highlights the clinical importance of pursuing further genetic evaluation in patients diagnosed with chromosome 6q24-related neonatal hyperglycemia via targeted methylation-specific multiplex ligation-dependent probe amplification analysis identifying a duplication in this region. Early identification and intervention can improve developmental outcomes for patients with larger chromosome 6q duplications. Oxford University Press 2020-04-27 /pmc/articles/PMC7185952/ /pubmed/32373772 http://dx.doi.org/10.1210/jendso/bvaa027 Text en Published by Oxford University Press on behalf of the Endocrine Society 2020 http://creativecommons.org/licenses/by-nc-nd/4.0/ This is an Open Access article distributed under the terms of the Creative Commons Attribution-NonCommercial-NoDerivs licence (http://creativecommons.org/licenses/by-nc-nd/4.0/), which permits non-commercial reproduction and distribution of the work, in any medium, provided the original work is not altered or transformed in any way, and that the work is properly cited. For commercial re-use, please contact journals.permissions@oup.com
spellingShingle Case Report
Gore, Rachel H
Nikita, Maria Eleni
Newton, Paula G
Carter, Rebecca G
Reyes-Bautista, Jeanine
Greene, Carol L
Duplication 6q24: More Than Just Diabetes
title Duplication 6q24: More Than Just Diabetes
title_full Duplication 6q24: More Than Just Diabetes
title_fullStr Duplication 6q24: More Than Just Diabetes
title_full_unstemmed Duplication 6q24: More Than Just Diabetes
title_short Duplication 6q24: More Than Just Diabetes
title_sort duplication 6q24: more than just diabetes
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7185952/
https://www.ncbi.nlm.nih.gov/pubmed/32373772
http://dx.doi.org/10.1210/jendso/bvaa027
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