Screening for obstructive sleep apnea in treacher-collins syndrome

Objectives/Hypothesis: This study evaluated the accuracy of established obstructive sleep apnea syndrome (OSAS) questionnaires based on presenting symptoms and complaints as screening tools for OSAS in Treacher‐Collins syndrome (TCS). Study Design: Cross‐sectional cohort study. Methods: In 35 TCS pa...

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Veröffentlicht in:The Laryngoscope 2012-04, Vol.122 (4), p.930-934
Hauptverfasser: Plomp, Raul G., Joosten, Koen F. M., Wolvius, Eppo B., Hoeve, Hans L. J., Poublon, Rene M. L., van Montfort, Kees A. G. M., Bredero-Boelhouwer, Hansje H., Mathijssen, Irene M. J.
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container_end_page 934
container_issue 4
container_start_page 930
container_title The Laryngoscope
container_volume 122
creator Plomp, Raul G.
Joosten, Koen F. M.
Wolvius, Eppo B.
Hoeve, Hans L. J.
Poublon, Rene M. L.
van Montfort, Kees A. G. M.
Bredero-Boelhouwer, Hansje H.
Mathijssen, Irene M. J.
description Objectives/Hypothesis: This study evaluated the accuracy of established obstructive sleep apnea syndrome (OSAS) questionnaires based on presenting symptoms and complaints as screening tools for OSAS in Treacher‐Collins syndrome (TCS). Study Design: Cross‐sectional cohort study. Methods: In 35 TCS patients (13 children, 22 adults) in whom diagnostic polysomnographic results on OSAS were available, the Brouillette score was evaluated in children and the Epworth Sleepiness Scale in adults. Results: The total Brouillette score showed a sensitivity of 50%, specificity of 71%, and positive and negative predictive values of 60% and 63%, respectively. The answer “No” to the question as to whether a child snored could rule out OSAS in children, and showed positive and negative predictive values of 55% and 100%, respectively. The Epworth Sleepiness Scale showed a sensitivity of 0%, specificity of 92%, and positive and negative predictive values of 0% and 57%, respectively. A positive answer to the question of whether a person falls asleep while sitting and talking to someone (sometimes or more) was able to predict OSAS in adults; this question had positive and negative predictive values of 100% and 72%, respectively. Conclusions: This cross‐sectional cohort study showed that the Brouillette score and the Epworth Sleepiness Scale are of minimal usefulness in TCS. Diagnosis of OSAS based solely on complaints is not reliable, probably due to habituation. Therefore, for a good evaluation and optimal multidisciplinary treatment of this chronic disease in TCS, all newly referred pediatric and adult TCS patients should be screened for OSAS at least once with polysomnography. Laryngoscope, 2012
doi_str_mv 10.1002/lary.23187
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M. ; Wolvius, Eppo B. ; Hoeve, Hans L. J. ; Poublon, Rene M. L. ; van Montfort, Kees A. G. M. ; Bredero-Boelhouwer, Hansje H. ; Mathijssen, Irene M. J.</creator><creatorcontrib>Plomp, Raul G. ; Joosten, Koen F. M. ; Wolvius, Eppo B. ; Hoeve, Hans L. J. ; Poublon, Rene M. L. ; van Montfort, Kees A. G. M. ; Bredero-Boelhouwer, Hansje H. ; Mathijssen, Irene M. J.</creatorcontrib><description>Objectives/Hypothesis: This study evaluated the accuracy of established obstructive sleep apnea syndrome (OSAS) questionnaires based on presenting symptoms and complaints as screening tools for OSAS in Treacher‐Collins syndrome (TCS). Study Design: Cross‐sectional cohort study. Methods: In 35 TCS patients (13 children, 22 adults) in whom diagnostic polysomnographic results on OSAS were available, the Brouillette score was evaluated in children and the Epworth Sleepiness Scale in adults. Results: The total Brouillette score showed a sensitivity of 50%, specificity of 71%, and positive and negative predictive values of 60% and 63%, respectively. The answer “No” to the question as to whether a child snored could rule out OSAS in children, and showed positive and negative predictive values of 55% and 100%, respectively. The Epworth Sleepiness Scale showed a sensitivity of 0%, specificity of 92%, and positive and negative predictive values of 0% and 57%, respectively. A positive answer to the question of whether a person falls asleep while sitting and talking to someone (sometimes or more) was able to predict OSAS in adults; this question had positive and negative predictive values of 100% and 72%, respectively. Conclusions: This cross‐sectional cohort study showed that the Brouillette score and the Epworth Sleepiness Scale are of minimal usefulness in TCS. Diagnosis of OSAS based solely on complaints is not reliable, probably due to habituation. Therefore, for a good evaluation and optimal multidisciplinary treatment of this chronic disease in TCS, all newly referred pediatric and adult TCS patients should be screened for OSAS at least once with polysomnography. Laryngoscope, 2012</description><identifier>ISSN: 0023-852X</identifier><identifier>EISSN: 1531-4995</identifier><identifier>DOI: 10.1002/lary.23187</identifier><identifier>PMID: 22374652</identifier><identifier>CODEN: LARYA8</identifier><language>eng</language><publisher>Hoboken: Wiley Subscription Services, Inc., A Wiley Company</publisher><subject>Adolescent ; Adult ; Biological and medical sciences ; Brouillette score ; Child ; Child, Preschool ; Cross-Sectional Studies ; Diseases of the osteoarticular system ; Epworth Sleepiness Scale ; Female ; Humans ; Incidence ; Infant ; Infant, Newborn ; Level of Evidence: 2b ; Male ; Malformations and congenital and or hereditary diseases involving bones. Joint deformations ; Mandibulofacial Dysostosis - complications ; Mandibulofacial Dysostosis - epidemiology ; Mass Screening - methods ; Medical sciences ; Middle Aged ; Netherlands - epidemiology ; obstructive sleep apnea ; Otorhinolaryngology. Stomatology ; Pneumology ; Polysomnography ; Prevalence ; Respiratory system : syndromes and miscellaneous diseases ; Retrospective Studies ; Risk Factors ; screening ; Sleep apnea ; Sleep Apnea, Obstructive - diagnosis ; Sleep Apnea, Obstructive - epidemiology ; Sleep Apnea, Obstructive - etiology ; Sleep disorders ; Surveys and Questionnaires ; Treacher-Collins syndrome ; Young Adult</subject><ispartof>The Laryngoscope, 2012-04, Vol.122 (4), p.930-934</ispartof><rights>Copyright © 2012 The American Laryngological, Rhinological, and Otological Society, Inc.</rights><rights>2015 INIST-CNRS</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c4247-857949ee61b8bb8fdd62dea8e0897e14b7ff01528723536503d87c40a14584883</citedby><cites>FETCH-LOGICAL-c4247-857949ee61b8bb8fdd62dea8e0897e14b7ff01528723536503d87c40a14584883</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://onlinelibrary.wiley.com/doi/pdf/10.1002%2Flary.23187$$EPDF$$P50$$Gwiley$$H</linktopdf><linktohtml>$$Uhttps://onlinelibrary.wiley.com/doi/full/10.1002%2Flary.23187$$EHTML$$P50$$Gwiley$$H</linktohtml><link.rule.ids>314,776,780,1411,27901,27902,45550,45551</link.rule.ids><backlink>$$Uhttp://pascal-francis.inist.fr/vibad/index.php?action=getRecordDetail&amp;idt=25698037$$DView record in Pascal Francis$$Hfree_for_read</backlink><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/22374652$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Plomp, Raul G.</creatorcontrib><creatorcontrib>Joosten, Koen F. M.</creatorcontrib><creatorcontrib>Wolvius, Eppo B.</creatorcontrib><creatorcontrib>Hoeve, Hans L. J.</creatorcontrib><creatorcontrib>Poublon, Rene M. L.</creatorcontrib><creatorcontrib>van Montfort, Kees A. G. M.</creatorcontrib><creatorcontrib>Bredero-Boelhouwer, Hansje H.</creatorcontrib><creatorcontrib>Mathijssen, Irene M. J.</creatorcontrib><title>Screening for obstructive sleep apnea in treacher-collins syndrome</title><title>The Laryngoscope</title><addtitle>The Laryngoscope</addtitle><description>Objectives/Hypothesis: This study evaluated the accuracy of established obstructive sleep apnea syndrome (OSAS) questionnaires based on presenting symptoms and complaints as screening tools for OSAS in Treacher‐Collins syndrome (TCS). Study Design: Cross‐sectional cohort study. Methods: In 35 TCS patients (13 children, 22 adults) in whom diagnostic polysomnographic results on OSAS were available, the Brouillette score was evaluated in children and the Epworth Sleepiness Scale in adults. Results: The total Brouillette score showed a sensitivity of 50%, specificity of 71%, and positive and negative predictive values of 60% and 63%, respectively. The answer “No” to the question as to whether a child snored could rule out OSAS in children, and showed positive and negative predictive values of 55% and 100%, respectively. The Epworth Sleepiness Scale showed a sensitivity of 0%, specificity of 92%, and positive and negative predictive values of 0% and 57%, respectively. A positive answer to the question of whether a person falls asleep while sitting and talking to someone (sometimes or more) was able to predict OSAS in adults; this question had positive and negative predictive values of 100% and 72%, respectively. Conclusions: This cross‐sectional cohort study showed that the Brouillette score and the Epworth Sleepiness Scale are of minimal usefulness in TCS. Diagnosis of OSAS based solely on complaints is not reliable, probably due to habituation. Therefore, for a good evaluation and optimal multidisciplinary treatment of this chronic disease in TCS, all newly referred pediatric and adult TCS patients should be screened for OSAS at least once with polysomnography. 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Joint deformations</subject><subject>Mandibulofacial Dysostosis - complications</subject><subject>Mandibulofacial Dysostosis - epidemiology</subject><subject>Mass Screening - methods</subject><subject>Medical sciences</subject><subject>Middle Aged</subject><subject>Netherlands - epidemiology</subject><subject>obstructive sleep apnea</subject><subject>Otorhinolaryngology. Stomatology</subject><subject>Pneumology</subject><subject>Polysomnography</subject><subject>Prevalence</subject><subject>Respiratory system : syndromes and miscellaneous diseases</subject><subject>Retrospective Studies</subject><subject>Risk Factors</subject><subject>screening</subject><subject>Sleep apnea</subject><subject>Sleep Apnea, Obstructive - diagnosis</subject><subject>Sleep Apnea, Obstructive - epidemiology</subject><subject>Sleep Apnea, Obstructive - etiology</subject><subject>Sleep disorders</subject><subject>Surveys and Questionnaires</subject><subject>Treacher-Collins syndrome</subject><subject>Young Adult</subject><issn>0023-852X</issn><issn>1531-4995</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2012</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNp9kN9PFDEQxxujgRN58Q8gmxhDYrLQn9vuIxI8NAcCapCnptud1WKve7a74v339rgDEx-cl3mYz3xn8kHoJcEHBGN66E1cHlBGlHyCJkQwUvK6Fk_RJA9ZqQT9uo2ep3SLMZFM4C20TSmTvBJ0gt5-shEguPCt6PpY9E0a4mgH9wuK5AEWhVkEMIULxRDB2O8QS9t770Iq0jK0sZ_DC_SsMz7B7qbvoC_vTj4fn5azj9P3x0ez0nLKZf5D1rwGqEijmkZ1bVvRFowCrGoJhDey6zARVEnKBKsEZq2SlmNDuFBcKbaD9te5i9j_HCENeu6SBe9NgH5MumacyiyEZfLVP-RtP8aQn9OEs1xYkhX1Zk3Z2KcUodOL6ObZpSZYr8TqlVh9LzbDe5vIsZlD-4g-mMzA6w1gkjW-iyZYl_5yoqoVZqsgsubunIflf07q2dHVzcPxcr3j0gC_H3dM_KEryaTQ1-dTTS7PLqYfri-zhz-ntJ2b</recordid><startdate>201204</startdate><enddate>201204</enddate><creator>Plomp, Raul G.</creator><creator>Joosten, Koen F. 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Joint deformations</topic><topic>Mandibulofacial Dysostosis - complications</topic><topic>Mandibulofacial Dysostosis - epidemiology</topic><topic>Mass Screening - methods</topic><topic>Medical sciences</topic><topic>Middle Aged</topic><topic>Netherlands - epidemiology</topic><topic>obstructive sleep apnea</topic><topic>Otorhinolaryngology. Stomatology</topic><topic>Pneumology</topic><topic>Polysomnography</topic><topic>Prevalence</topic><topic>Respiratory system : syndromes and miscellaneous diseases</topic><topic>Retrospective Studies</topic><topic>Risk Factors</topic><topic>screening</topic><topic>Sleep apnea</topic><topic>Sleep Apnea, Obstructive - diagnosis</topic><topic>Sleep Apnea, Obstructive - epidemiology</topic><topic>Sleep Apnea, Obstructive - etiology</topic><topic>Sleep disorders</topic><topic>Surveys and Questionnaires</topic><topic>Treacher-Collins syndrome</topic><topic>Young Adult</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Plomp, Raul G.</creatorcontrib><creatorcontrib>Joosten, Koen F. M.</creatorcontrib><creatorcontrib>Wolvius, Eppo B.</creatorcontrib><creatorcontrib>Hoeve, Hans L. J.</creatorcontrib><creatorcontrib>Poublon, Rene M. L.</creatorcontrib><creatorcontrib>van Montfort, Kees A. G. M.</creatorcontrib><creatorcontrib>Bredero-Boelhouwer, Hansje H.</creatorcontrib><creatorcontrib>Mathijssen, Irene M. J.</creatorcontrib><collection>Istex</collection><collection>Pascal-Francis</collection><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>ProQuest Health &amp; Medical Complete (Alumni)</collection><collection>MEDLINE - Academic</collection><jtitle>The Laryngoscope</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Plomp, Raul G.</au><au>Joosten, Koen F. M.</au><au>Wolvius, Eppo B.</au><au>Hoeve, Hans L. J.</au><au>Poublon, Rene M. L.</au><au>van Montfort, Kees A. G. M.</au><au>Bredero-Boelhouwer, Hansje H.</au><au>Mathijssen, Irene M. J.</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Screening for obstructive sleep apnea in treacher-collins syndrome</atitle><jtitle>The Laryngoscope</jtitle><addtitle>The Laryngoscope</addtitle><date>2012-04</date><risdate>2012</risdate><volume>122</volume><issue>4</issue><spage>930</spage><epage>934</epage><pages>930-934</pages><issn>0023-852X</issn><eissn>1531-4995</eissn><coden>LARYA8</coden><abstract>Objectives/Hypothesis: This study evaluated the accuracy of established obstructive sleep apnea syndrome (OSAS) questionnaires based on presenting symptoms and complaints as screening tools for OSAS in Treacher‐Collins syndrome (TCS). Study Design: Cross‐sectional cohort study. Methods: In 35 TCS patients (13 children, 22 adults) in whom diagnostic polysomnographic results on OSAS were available, the Brouillette score was evaluated in children and the Epworth Sleepiness Scale in adults. Results: The total Brouillette score showed a sensitivity of 50%, specificity of 71%, and positive and negative predictive values of 60% and 63%, respectively. The answer “No” to the question as to whether a child snored could rule out OSAS in children, and showed positive and negative predictive values of 55% and 100%, respectively. The Epworth Sleepiness Scale showed a sensitivity of 0%, specificity of 92%, and positive and negative predictive values of 0% and 57%, respectively. A positive answer to the question of whether a person falls asleep while sitting and talking to someone (sometimes or more) was able to predict OSAS in adults; this question had positive and negative predictive values of 100% and 72%, respectively. Conclusions: This cross‐sectional cohort study showed that the Brouillette score and the Epworth Sleepiness Scale are of minimal usefulness in TCS. Diagnosis of OSAS based solely on complaints is not reliable, probably due to habituation. Therefore, for a good evaluation and optimal multidisciplinary treatment of this chronic disease in TCS, all newly referred pediatric and adult TCS patients should be screened for OSAS at least once with polysomnography. Laryngoscope, 2012</abstract><cop>Hoboken</cop><pub>Wiley Subscription Services, Inc., A Wiley Company</pub><pmid>22374652</pmid><doi>10.1002/lary.23187</doi><tpages>5</tpages></addata></record>
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subjects Adolescent
Adult
Biological and medical sciences
Brouillette score
Child
Child, Preschool
Cross-Sectional Studies
Diseases of the osteoarticular system
Epworth Sleepiness Scale
Female
Humans
Incidence
Infant
Infant, Newborn
Level of Evidence: 2b
Male
Malformations and congenital and or hereditary diseases involving bones. Joint deformations
Mandibulofacial Dysostosis - complications
Mandibulofacial Dysostosis - epidemiology
Mass Screening - methods
Medical sciences
Middle Aged
Netherlands - epidemiology
obstructive sleep apnea
Otorhinolaryngology. Stomatology
Pneumology
Polysomnography
Prevalence
Respiratory system : syndromes and miscellaneous diseases
Retrospective Studies
Risk Factors
screening
Sleep apnea
Sleep Apnea, Obstructive - diagnosis
Sleep Apnea, Obstructive - epidemiology
Sleep Apnea, Obstructive - etiology
Sleep disorders
Surveys and Questionnaires
Treacher-Collins syndrome
Young Adult
title Screening for obstructive sleep apnea in treacher-collins syndrome
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