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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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Published in:The Laryngoscope 2021-12, Vol.131 (12), p.2811-2816
Main Authors: 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.
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cited_by cdi_FETCH-LOGICAL-c3345-2046fa60b0a4d16183b27df76686d401d87a8e205b08ae3b9740cd380c4c94983
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container_end_page 2816
container_issue 12
container_start_page 2811
container_title The Laryngoscope
container_volume 131
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
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C. ; Zanin, Eduardo M. ; Portinho, Ciro P. ; Collares, Marcus V. M.</creator><creatorcontrib>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.</creatorcontrib><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. 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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. 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C.</creatorcontrib><creatorcontrib>Zanin, Eduardo M.</creatorcontrib><creatorcontrib>Portinho, Ciro P.</creatorcontrib><creatorcontrib>Collares, Marcus V. M.</creatorcontrib><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>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 &amp; 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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