Predictors of Respiratory Dysfunction at Diagnosis of Robin Sequence
Objectives/Hypothesis Robin sequence (RS) consists of associated micrognathia, glossoptosis, and respiratory dysfunction, with or without cleft palate. Studies on how different patient characteristics impact the severity of respiratory dysfunction are scarce and contradictory; this study investigate...
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Veröffentlicht in: | The Laryngoscope 2021-12, Vol.131 (12), p.2811-2816 |
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creator | Duarte, Daniele W. Schweiger, Claudia Manica, Denise Fagondes, Simone Fleith, Iran J. Rojas, Juan J. C. Zanin, Eduardo M. Portinho, Ciro P. Collares, Marcus V. M. |
description | Objectives/Hypothesis
Robin sequence (RS) consists of associated micrognathia, glossoptosis, and respiratory dysfunction, with or without cleft palate. Studies on how different patient characteristics impact the severity of respiratory dysfunction are scarce and contradictory; this study investigates how different features affect respiratory obstruction severity at diagnosis of RS in controlled analysis.
Study Design
Retrospective cohort study that enrolled 71 RS patients under 90 days old who received care in our institution from 2009 to 2020.
Methods
The primary outcome, respiratory dysfunction, was categorized into four severity groups and analyzed using a multinomial logistic regression model that considered age, sex, mandible length, cleft palate, syndromic diagnosis, other airway anomalies, and degree of glossoptosis.
Results
Mandible length, syndromic diagnosis, and Yellon grade 3 glossoptosis were related to poorer respiratory outcomes (need for respiratory support). In univariate analysis, for each additional 1 mm of mandible length at diagnosis, a mean reduction of 28% in the risk of needing respiratory support was observed (OR = 0.72; 0.58–0.89); syndromic diagnosis and grade 3 glossoptosis also raised the risk (OR = 6.50; 1.59–26.51 and OR = 12.75; 1.03–157.14, respectively). In multivariate analysis, only mandible length significantly maintained its effects (OR = 0.73; 0.56–0.96), a 27% reduction.
Conclusions
Mandible length was an independent predictor for more severe respiratory dysfunction in RS patients, with larger mandibles showing protective effects. Syndromic diagnosis and Yellon grade 3 glossoptosis are also likely to be associated with poorer respiratory outcomes, although this was not demonstrated in multivariate analysis.
Level of Evidence
4 Laryngoscope, 131:2811–2816, 2021 |
doi_str_mv | 10.1002/lary.29685 |
format | Article |
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Robin sequence (RS) consists of associated micrognathia, glossoptosis, and respiratory dysfunction, with or without cleft palate. Studies on how different patient characteristics impact the severity of respiratory dysfunction are scarce and contradictory; this study investigates how different features affect respiratory obstruction severity at diagnosis of RS in controlled analysis.
Study Design
Retrospective cohort study that enrolled 71 RS patients under 90 days old who received care in our institution from 2009 to 2020.
Methods
The primary outcome, respiratory dysfunction, was categorized into four severity groups and analyzed using a multinomial logistic regression model that considered age, sex, mandible length, cleft palate, syndromic diagnosis, other airway anomalies, and degree of glossoptosis.
Results
Mandible length, syndromic diagnosis, and Yellon grade 3 glossoptosis were related to poorer respiratory outcomes (need for respiratory support). In univariate analysis, for each additional 1 mm of mandible length at diagnosis, a mean reduction of 28% in the risk of needing respiratory support was observed (OR = 0.72; 0.58–0.89); syndromic diagnosis and grade 3 glossoptosis also raised the risk (OR = 6.50; 1.59–26.51 and OR = 12.75; 1.03–157.14, respectively). In multivariate analysis, only mandible length significantly maintained its effects (OR = 0.73; 0.56–0.96), a 27% reduction.
Conclusions
Mandible length was an independent predictor for more severe respiratory dysfunction in RS patients, with larger mandibles showing protective effects. Syndromic diagnosis and Yellon grade 3 glossoptosis are also likely to be associated with poorer respiratory outcomes, although this was not demonstrated in multivariate analysis.
Level of Evidence
4 Laryngoscope, 131:2811–2816, 2021</description><identifier>ISSN: 0023-852X</identifier><identifier>EISSN: 1531-4995</identifier><identifier>DOI: 10.1002/lary.29685</identifier><language>eng</language><publisher>Hoboken, USA: John Wiley & Sons, Inc</publisher><subject>Babies ; Birth defects ; Laryngoscopy ; mandible ; Multivariate analysis ; obstructive sleep apnea ; respiratory dysfunction ; Robin sequence ; Sleep apnea</subject><ispartof>The Laryngoscope, 2021-12, Vol.131 (12), p.2811-2816</ispartof><rights>2021 The American Laryngological, Rhinological and Otological Society, Inc..</rights><rights>2021 The American Laryngological, Rhinological and Otological Society, Inc.</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c3345-2046fa60b0a4d16183b27df76686d401d87a8e205b08ae3b9740cd380c4c94983</citedby><cites>FETCH-LOGICAL-c3345-2046fa60b0a4d16183b27df76686d401d87a8e205b08ae3b9740cd380c4c94983</cites><orcidid>0000-0002-2438-3355 ; 0000-0002-1190-3390</orcidid></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.29685$$EPDF$$P50$$Gwiley$$H</linktopdf><linktohtml>$$Uhttps://onlinelibrary.wiley.com/doi/full/10.1002%2Flary.29685$$EHTML$$P50$$Gwiley$$H</linktohtml><link.rule.ids>314,780,784,1417,27924,27925,45574,45575</link.rule.ids></links><search><creatorcontrib>Duarte, Daniele W.</creatorcontrib><creatorcontrib>Schweiger, Claudia</creatorcontrib><creatorcontrib>Manica, Denise</creatorcontrib><creatorcontrib>Fagondes, Simone</creatorcontrib><creatorcontrib>Fleith, Iran J.</creatorcontrib><creatorcontrib>Rojas, Juan J. C.</creatorcontrib><creatorcontrib>Zanin, Eduardo M.</creatorcontrib><creatorcontrib>Portinho, Ciro P.</creatorcontrib><creatorcontrib>Collares, Marcus V. M.</creatorcontrib><title>Predictors of Respiratory Dysfunction at Diagnosis of Robin Sequence</title><title>The Laryngoscope</title><description>Objectives/Hypothesis
Robin sequence (RS) consists of associated micrognathia, glossoptosis, and respiratory dysfunction, with or without cleft palate. Studies on how different patient characteristics impact the severity of respiratory dysfunction are scarce and contradictory; this study investigates how different features affect respiratory obstruction severity at diagnosis of RS in controlled analysis.
Study Design
Retrospective cohort study that enrolled 71 RS patients under 90 days old who received care in our institution from 2009 to 2020.
Methods
The primary outcome, respiratory dysfunction, was categorized into four severity groups and analyzed using a multinomial logistic regression model that considered age, sex, mandible length, cleft palate, syndromic diagnosis, other airway anomalies, and degree of glossoptosis.
Results
Mandible length, syndromic diagnosis, and Yellon grade 3 glossoptosis were related to poorer respiratory outcomes (need for respiratory support). In univariate analysis, for each additional 1 mm of mandible length at diagnosis, a mean reduction of 28% in the risk of needing respiratory support was observed (OR = 0.72; 0.58–0.89); syndromic diagnosis and grade 3 glossoptosis also raised the risk (OR = 6.50; 1.59–26.51 and OR = 12.75; 1.03–157.14, respectively). In multivariate analysis, only mandible length significantly maintained its effects (OR = 0.73; 0.56–0.96), a 27% reduction.
Conclusions
Mandible length was an independent predictor for more severe respiratory dysfunction in RS patients, with larger mandibles showing protective effects. Syndromic diagnosis and Yellon grade 3 glossoptosis are also likely to be associated with poorer respiratory outcomes, although this was not demonstrated in multivariate analysis.
Level of Evidence
4 Laryngoscope, 131:2811–2816, 2021</description><subject>Babies</subject><subject>Birth defects</subject><subject>Laryngoscopy</subject><subject>mandible</subject><subject>Multivariate analysis</subject><subject>obstructive sleep apnea</subject><subject>respiratory dysfunction</subject><subject>Robin sequence</subject><subject>Sleep apnea</subject><issn>0023-852X</issn><issn>1531-4995</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2021</creationdate><recordtype>article</recordtype><recordid>eNp90E1LwzAYB_AgCs7pxU9Q8CJC55PmpclxbL7BQJkKeippmkpG18ykRfrtzawnD57CQ37PC3-EzjHMMEB23Sg_zDLJBTtAE8wITqmU7BBN4idJBcvejtFJCBsAnBMGE7R88qayunM-JK5O1ibsrFexHJLlEOq-1Z11baK6ZGnVR-uCHZ0rbZs8m8_etNqcoqNaNcGc_b5T9Hp787K4T1ePdw-L-SrVhFCWZkB5rTiUoGiFORakzPKqzjkXvKKAK5ErYTJgJQhlSClzCroiAjTVkkpBpuhynLvzLm4OXbG1QZumUa1xfSgyRoFhLLiM9OIP3bjet_G6qCSTIlIS1dWotHcheFMXO2-3McMCQ7EPtNgHWvwEGjEe8ZdtzPCPLFbz9fvY8w1MKHcC</recordid><startdate>202112</startdate><enddate>202112</enddate><creator>Duarte, Daniele W.</creator><creator>Schweiger, Claudia</creator><creator>Manica, Denise</creator><creator>Fagondes, Simone</creator><creator>Fleith, Iran J.</creator><creator>Rojas, Juan J. C.</creator><creator>Zanin, Eduardo M.</creator><creator>Portinho, Ciro P.</creator><creator>Collares, Marcus V. M.</creator><general>John Wiley & Sons, Inc</general><general>Wiley Subscription Services, Inc</general><scope>AAYXX</scope><scope>CITATION</scope><scope>K9.</scope><scope>7X8</scope><orcidid>https://orcid.org/0000-0002-2438-3355</orcidid><orcidid>https://orcid.org/0000-0002-1190-3390</orcidid></search><sort><creationdate>202112</creationdate><title>Predictors of Respiratory Dysfunction at Diagnosis of Robin Sequence</title><author>Duarte, Daniele W. ; Schweiger, Claudia ; Manica, Denise ; Fagondes, Simone ; Fleith, Iran J. ; Rojas, Juan J. C. ; Zanin, Eduardo M. ; Portinho, Ciro P. ; Collares, Marcus V. M.</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c3345-2046fa60b0a4d16183b27df76686d401d87a8e205b08ae3b9740cd380c4c94983</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2021</creationdate><topic>Babies</topic><topic>Birth defects</topic><topic>Laryngoscopy</topic><topic>mandible</topic><topic>Multivariate analysis</topic><topic>obstructive sleep apnea</topic><topic>respiratory dysfunction</topic><topic>Robin sequence</topic><topic>Sleep apnea</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Duarte, Daniele W.</creatorcontrib><creatorcontrib>Schweiger, Claudia</creatorcontrib><creatorcontrib>Manica, Denise</creatorcontrib><creatorcontrib>Fagondes, Simone</creatorcontrib><creatorcontrib>Fleith, Iran J.</creatorcontrib><creatorcontrib>Rojas, Juan J. C.</creatorcontrib><creatorcontrib>Zanin, Eduardo M.</creatorcontrib><creatorcontrib>Portinho, Ciro P.</creatorcontrib><creatorcontrib>Collares, Marcus V. M.</creatorcontrib><collection>CrossRef</collection><collection>ProQuest Health & 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>Duarte, Daniele W.</au><au>Schweiger, Claudia</au><au>Manica, Denise</au><au>Fagondes, Simone</au><au>Fleith, Iran J.</au><au>Rojas, Juan J. C.</au><au>Zanin, Eduardo M.</au><au>Portinho, Ciro P.</au><au>Collares, Marcus V. M.</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Predictors of Respiratory Dysfunction at Diagnosis of Robin Sequence</atitle><jtitle>The Laryngoscope</jtitle><date>2021-12</date><risdate>2021</risdate><volume>131</volume><issue>12</issue><spage>2811</spage><epage>2816</epage><pages>2811-2816</pages><issn>0023-852X</issn><eissn>1531-4995</eissn><abstract>Objectives/Hypothesis
Robin sequence (RS) consists of associated micrognathia, glossoptosis, and respiratory dysfunction, with or without cleft palate. Studies on how different patient characteristics impact the severity of respiratory dysfunction are scarce and contradictory; this study investigates how different features affect respiratory obstruction severity at diagnosis of RS in controlled analysis.
Study Design
Retrospective cohort study that enrolled 71 RS patients under 90 days old who received care in our institution from 2009 to 2020.
Methods
The primary outcome, respiratory dysfunction, was categorized into four severity groups and analyzed using a multinomial logistic regression model that considered age, sex, mandible length, cleft palate, syndromic diagnosis, other airway anomalies, and degree of glossoptosis.
Results
Mandible length, syndromic diagnosis, and Yellon grade 3 glossoptosis were related to poorer respiratory outcomes (need for respiratory support). In univariate analysis, for each additional 1 mm of mandible length at diagnosis, a mean reduction of 28% in the risk of needing respiratory support was observed (OR = 0.72; 0.58–0.89); syndromic diagnosis and grade 3 glossoptosis also raised the risk (OR = 6.50; 1.59–26.51 and OR = 12.75; 1.03–157.14, respectively). In multivariate analysis, only mandible length significantly maintained its effects (OR = 0.73; 0.56–0.96), a 27% reduction.
Conclusions
Mandible length was an independent predictor for more severe respiratory dysfunction in RS patients, with larger mandibles showing protective effects. Syndromic diagnosis and Yellon grade 3 glossoptosis are also likely to be associated with poorer respiratory outcomes, although this was not demonstrated in multivariate analysis.
Level of Evidence
4 Laryngoscope, 131:2811–2816, 2021</abstract><cop>Hoboken, USA</cop><pub>John Wiley & Sons, Inc</pub><doi>10.1002/lary.29685</doi><tpages>6</tpages><orcidid>https://orcid.org/0000-0002-2438-3355</orcidid><orcidid>https://orcid.org/0000-0002-1190-3390</orcidid></addata></record> |
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subjects | Babies Birth defects Laryngoscopy mandible Multivariate analysis obstructive sleep apnea respiratory dysfunction Robin sequence Sleep apnea |
title | Predictors of Respiratory Dysfunction at Diagnosis of Robin Sequence |
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