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Growth charts for Thai children with Prader-Willi syndrome aged 0–18 years

BACKGROUND: Prader-Willi syndrome (PWS) is a multisystem genetic disorder, which has a typical eating behavior and growth pattern. In the infancy period, children with PWS have low body weight followed by hyperphagia in later childhood. Disease-specific growth charts have been recommended for monito...

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Autores principales: Mongkollarp, Nantiya, Tim-Aroon, Thipwimol, Okascharoen, Chusak, Wichajarn, Khunton, Phosuwattanakul, Jeeraparn, Chongviriyaphan, Nalinee, Wattanasirichaigoon, Duangrurdee
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2020
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7201534/
https://www.ncbi.nlm.nih.gov/pubmed/32375863
http://dx.doi.org/10.1186/s13023-020-01388-7
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author Mongkollarp, Nantiya
Tim-Aroon, Thipwimol
Okascharoen, Chusak
Wichajarn, Khunton
Phosuwattanakul, Jeeraparn
Chongviriyaphan, Nalinee
Wattanasirichaigoon, Duangrurdee
author_facet Mongkollarp, Nantiya
Tim-Aroon, Thipwimol
Okascharoen, Chusak
Wichajarn, Khunton
Phosuwattanakul, Jeeraparn
Chongviriyaphan, Nalinee
Wattanasirichaigoon, Duangrurdee
author_sort Mongkollarp, Nantiya
collection PubMed
description BACKGROUND: Prader-Willi syndrome (PWS) is a multisystem genetic disorder, which has a typical eating behavior and growth pattern. In the infancy period, children with PWS have low body weight followed by hyperphagia in later childhood. Disease-specific growth charts have been recommended for monitoring PWS patients. Previous literature demonstrated growth differences among individuals with PWS of different ethnicity. METHODS: A retrospective multicenter study was performed in PWS patients from different areas of Thailand included collaboration with the Thai PWS support group during 2000–2017. Baseline characteristics and anthropometric data were reviewed. Both growth hormone and non-growth hormone received patients were included, but the data after receiving GH were excluded before curve construction. Growth charts for Thai PWS compared to the 50th normative centile were constructed using Generalized Least Squares (GLS) methods. Curve smoothing was performed by Fractional Polynomials and Exponential Transformation. RESULT: One hundred and thirteen patients with genetically confirmed PWS (55 males and 58 females) were enrolled. Fifty percent of patients were diagnosed less than 6 months of age. We developed growth charts for non-growth hormone treated Thai children with PWS aged between 0 and 18 years. A growth pattern was similar to other ethnicities while there were some differences. Mean birth weight of PWS patients was less than that of typical newborns. Mean adult height at 18 years of age in Thai children with PWS was lower than that in American children, but taller than Japanese. Mean weight of Thai PWS males at 18 years of age was more than those from other countries. CONCLUSION: This study is the first to document PWS-specific growth charts in Southeast Asian population. These growth charts will be useful in improving the quality of patient care and in evaluating the impact of growth hormone treatment in the future.
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spelling pubmed-72015342020-05-08 Growth charts for Thai children with Prader-Willi syndrome aged 0–18 years Mongkollarp, Nantiya Tim-Aroon, Thipwimol Okascharoen, Chusak Wichajarn, Khunton Phosuwattanakul, Jeeraparn Chongviriyaphan, Nalinee Wattanasirichaigoon, Duangrurdee Orphanet J Rare Dis Research BACKGROUND: Prader-Willi syndrome (PWS) is a multisystem genetic disorder, which has a typical eating behavior and growth pattern. In the infancy period, children with PWS have low body weight followed by hyperphagia in later childhood. Disease-specific growth charts have been recommended for monitoring PWS patients. Previous literature demonstrated growth differences among individuals with PWS of different ethnicity. METHODS: A retrospective multicenter study was performed in PWS patients from different areas of Thailand included collaboration with the Thai PWS support group during 2000–2017. Baseline characteristics and anthropometric data were reviewed. Both growth hormone and non-growth hormone received patients were included, but the data after receiving GH were excluded before curve construction. Growth charts for Thai PWS compared to the 50th normative centile were constructed using Generalized Least Squares (GLS) methods. Curve smoothing was performed by Fractional Polynomials and Exponential Transformation. RESULT: One hundred and thirteen patients with genetically confirmed PWS (55 males and 58 females) were enrolled. Fifty percent of patients were diagnosed less than 6 months of age. We developed growth charts for non-growth hormone treated Thai children with PWS aged between 0 and 18 years. A growth pattern was similar to other ethnicities while there were some differences. Mean birth weight of PWS patients was less than that of typical newborns. Mean adult height at 18 years of age in Thai children with PWS was lower than that in American children, but taller than Japanese. Mean weight of Thai PWS males at 18 years of age was more than those from other countries. CONCLUSION: This study is the first to document PWS-specific growth charts in Southeast Asian population. These growth charts will be useful in improving the quality of patient care and in evaluating the impact of growth hormone treatment in the future. BioMed Central 2020-05-06 /pmc/articles/PMC7201534/ /pubmed/32375863 http://dx.doi.org/10.1186/s13023-020-01388-7 Text en © The Author(s) 2020 Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated in a credit line to the data.
spellingShingle Research
Mongkollarp, Nantiya
Tim-Aroon, Thipwimol
Okascharoen, Chusak
Wichajarn, Khunton
Phosuwattanakul, Jeeraparn
Chongviriyaphan, Nalinee
Wattanasirichaigoon, Duangrurdee
Growth charts for Thai children with Prader-Willi syndrome aged 0–18 years
title Growth charts for Thai children with Prader-Willi syndrome aged 0–18 years
title_full Growth charts for Thai children with Prader-Willi syndrome aged 0–18 years
title_fullStr Growth charts for Thai children with Prader-Willi syndrome aged 0–18 years
title_full_unstemmed Growth charts for Thai children with Prader-Willi syndrome aged 0–18 years
title_short Growth charts for Thai children with Prader-Willi syndrome aged 0–18 years
title_sort growth charts for thai children with prader-willi syndrome aged 0–18 years
topic Research
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7201534/
https://www.ncbi.nlm.nih.gov/pubmed/32375863
http://dx.doi.org/10.1186/s13023-020-01388-7
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