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Gastro-oesophageal reflux - an important causative factor of severe tooth wear in Prader-Willi syndrome?

BACKGROUND: Prader-Willi syndrome (PWS) is the most common genetic human obesity syndrome and is characterized by hypotonia, endocrine disturbances, hyperphagia, obesity and mild mental retardation. Oral abnormalities, such as decreased salivary flow rates and extreme tooth wear, have also been desc...

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Autores principales: Saeves, Ronnaug, Strøm, Finn, Sandvik, Leiv, Nordgarden, Hilde
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2018
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5913865/
https://www.ncbi.nlm.nih.gov/pubmed/29685165
http://dx.doi.org/10.1186/s13023-018-0809-3
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author Saeves, Ronnaug
Strøm, Finn
Sandvik, Leiv
Nordgarden, Hilde
author_facet Saeves, Ronnaug
Strøm, Finn
Sandvik, Leiv
Nordgarden, Hilde
author_sort Saeves, Ronnaug
collection PubMed
description BACKGROUND: Prader-Willi syndrome (PWS) is the most common genetic human obesity syndrome and is characterized by hypotonia, endocrine disturbances, hyperphagia, obesity and mild mental retardation. Oral abnormalities, such as decreased salivary flow rates and extreme tooth wear, have also been described. Studies have shown a significant increase in reflux symptoms in individuals with obstuctive sleep apnoea syndrome and increased BMI, both of which are typical findings in PWS. Gastro-oesophageal reflux disease (GORD) has been identified in some individuals with PWS and is a significant intrinsic factor in dental tooth wear. The aim of this study was therefore to estimate the prevalence of GORD in adults and children and to evaluate a possible correlation between GORD and tooth wear in adults with PWS. They were all registered at the TAKO-centre. RESULTS: Twenty-nine individuals, 17 adults with a mean age of 32.6 years (range 18–48) and 12 children with a mean age of 8.8 years (range 3–17), agreed to undergo 24-hour oesophageal pH monitoring, and 90% of those enrolled managed to complete the examination. Four children and eleven adults were diagnosed with pathological gastro-oesophageal reflux, which is defined as acid exposure (pH less than 4) more than 3.6 or 4.3 percent of the time, respectively. Manometry performed in the adult group showed a pathologically high lower oesophageal sphincter pressure in four of the five individuals who had normal oesophageal pH values (pH under 4 less than 4.3% of the time). The two groups (reflux and non-reflux) were well balanced according to BMI, genotype, tooth grinding and hyposalivation. However, twice as many individuals in the reflux group as in the non-reflux group reported high consumption of acidic foods and drinks. Increased tooth wear was significantly correlated with GORD in the two groups (reflux n=6 and non-reflux n=6). CONCLUSIONS: The prevalence of gastro-oesophageal reflux is high in individuals with PWS. Tooth wear was strongly associated with GORD and acidic drinks, and both may be important aetiological factors underlying the extreme tooth wear in this group. Our data suggest a need for routine screening for GORD and dental wear in young individuals with Prader-Willi syndrome.
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spelling pubmed-59138652018-04-30 Gastro-oesophageal reflux - an important causative factor of severe tooth wear in Prader-Willi syndrome? Saeves, Ronnaug Strøm, Finn Sandvik, Leiv Nordgarden, Hilde Orphanet J Rare Dis Research BACKGROUND: Prader-Willi syndrome (PWS) is the most common genetic human obesity syndrome and is characterized by hypotonia, endocrine disturbances, hyperphagia, obesity and mild mental retardation. Oral abnormalities, such as decreased salivary flow rates and extreme tooth wear, have also been described. Studies have shown a significant increase in reflux symptoms in individuals with obstuctive sleep apnoea syndrome and increased BMI, both of which are typical findings in PWS. Gastro-oesophageal reflux disease (GORD) has been identified in some individuals with PWS and is a significant intrinsic factor in dental tooth wear. The aim of this study was therefore to estimate the prevalence of GORD in adults and children and to evaluate a possible correlation between GORD and tooth wear in adults with PWS. They were all registered at the TAKO-centre. RESULTS: Twenty-nine individuals, 17 adults with a mean age of 32.6 years (range 18–48) and 12 children with a mean age of 8.8 years (range 3–17), agreed to undergo 24-hour oesophageal pH monitoring, and 90% of those enrolled managed to complete the examination. Four children and eleven adults were diagnosed with pathological gastro-oesophageal reflux, which is defined as acid exposure (pH less than 4) more than 3.6 or 4.3 percent of the time, respectively. Manometry performed in the adult group showed a pathologically high lower oesophageal sphincter pressure in four of the five individuals who had normal oesophageal pH values (pH under 4 less than 4.3% of the time). The two groups (reflux and non-reflux) were well balanced according to BMI, genotype, tooth grinding and hyposalivation. However, twice as many individuals in the reflux group as in the non-reflux group reported high consumption of acidic foods and drinks. Increased tooth wear was significantly correlated with GORD in the two groups (reflux n=6 and non-reflux n=6). CONCLUSIONS: The prevalence of gastro-oesophageal reflux is high in individuals with PWS. Tooth wear was strongly associated with GORD and acidic drinks, and both may be important aetiological factors underlying the extreme tooth wear in this group. Our data suggest a need for routine screening for GORD and dental wear in young individuals with Prader-Willi syndrome. BioMed Central 2018-04-23 /pmc/articles/PMC5913865/ /pubmed/29685165 http://dx.doi.org/10.1186/s13023-018-0809-3 Text en © The Author(s). 2018 Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. 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.
spellingShingle Research
Saeves, Ronnaug
Strøm, Finn
Sandvik, Leiv
Nordgarden, Hilde
Gastro-oesophageal reflux - an important causative factor of severe tooth wear in Prader-Willi syndrome?
title Gastro-oesophageal reflux - an important causative factor of severe tooth wear in Prader-Willi syndrome?
title_full Gastro-oesophageal reflux - an important causative factor of severe tooth wear in Prader-Willi syndrome?
title_fullStr Gastro-oesophageal reflux - an important causative factor of severe tooth wear in Prader-Willi syndrome?
title_full_unstemmed Gastro-oesophageal reflux - an important causative factor of severe tooth wear in Prader-Willi syndrome?
title_short Gastro-oesophageal reflux - an important causative factor of severe tooth wear in Prader-Willi syndrome?
title_sort gastro-oesophageal reflux - an important causative factor of severe tooth wear in prader-willi syndrome?
topic Research
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5913865/
https://www.ncbi.nlm.nih.gov/pubmed/29685165
http://dx.doi.org/10.1186/s13023-018-0809-3
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