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Longitudinal evaluation of sleep disordered breathing in infants with Prader-Willi syndrome
ObjectiveTo evaluate the course of sleep disordered breathing (SDB) in infants with Prader-Willi syndrome (PWS).DesignRetrospective longitudinal observational study.SettingSleep laboratory at The Hospital for Sick Children, Toronto, Canada.PatientsInfants with PWS.Main outcome measuresThe natural hi...
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| Published in: | Archives of disease in childhood 2017-07, Vol.102 (7), p.638-638 |
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| container_title | Archives of disease in childhood |
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| creator | Khayat, Abdullah Narang, Indra Bin-Hasan, Saadoun Amin, Reshma Al-Saleh, Suhail |
| description | ObjectiveTo evaluate the course of sleep disordered breathing (SDB) in infants with Prader-Willi syndrome (PWS).DesignRetrospective longitudinal observational study.SettingSleep laboratory at The Hospital for Sick Children, Toronto, Canada.PatientsInfants with PWS.Main outcome measuresThe natural history of SDB in infants with PWS within 2 years from baseline assessment.ResultsWe identified 28 (12 male) infants with PWS who had a baseline polysomnography (PSG) at a median age (interquartile (IQR)) of 0.9 (0.5, 1.1) years. The median central apnoea index (CAI) at baseline was 6.6 events/hour (IQR 2.6, 12.1). Of these, 15/28 (53%) infants with PWS were diagnosed with significant central sleep apnoea (CSA) (CAI≥5 events/hour). Median age (IQR) at follow-up PSG was 2.1 (1.5, 2.6) years. The median CAI improved from 6.6 to 2.3 events/hour (p |
| doi_str_mv | 10.1136/archdischild-2016-311298 |
| format | article |
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The median central apnoea index (CAI) at baseline was 6.6 events/hour (IQR 2.6, 12.1). Of these, 15/28 (53%) infants with PWS were diagnosed with significant central sleep apnoea (CSA) (CAI≥5 events/hour). Median age (IQR) at follow-up PSG was 2.1 (1.5, 2.6) years. The median CAI improved from 6.6 to 2.3 events/hour (p<0.0001). Only four infants with PWS had persistent CSA at the time of the follow-up PSG. Furthermore, three infants with PWS were diagnosed with mild-to-moderate obstructive sleep apnoea (OSA) that has improved at follow-up studies whereas two patients with PWS with no evidence of OSA at baseline were diagnosed with severe OSA on the follow-up PSG requiring adenotonsillectomy. The overall median obstructive apnoea–hypopnoea index was similar between baseline and follow-up studies (0.6 and 0.8, respectively, p=0.91).ConclusionsCSA is prevalent in infants with PWS but usually improves with age. However, these patients continue to require ongoing PSG surveillance because some infants will have persistent CSA and others are at risk of developing OSA.</description><identifier>ISSN: 0003-9888</identifier><identifier>EISSN: 1468-2044</identifier><identifier>DOI: 10.1136/archdischild-2016-311298</identifier><identifier>PMID: 28130217</identifier><language>eng</language><publisher>England: BMJ Publishing Group Ltd</publisher><subject>Age ; Apnea ; Care and treatment ; Child, Preschool ; Children ; Endocrine therapy ; Female ; Genetic Disorders ; Growth disorders ; Growth hormones ; Health aspects ; Hospitals ; Humans ; Infant ; Infants ; Longitudinal Studies ; Male ; Observational studies ; Oxygen therapy ; Pediatrics ; Polysomnography ; Prader-Willi syndrome ; Prader-Willi Syndrome - complications ; Prospective Studies ; Respiration ; Sleep ; Sleep apnea ; Sleep Apnea Syndromes - etiology ; Sleep Apnea, Central - etiology ; Sleep deprivation ; Sleep disorders ; Young Children</subject><ispartof>Archives of disease in childhood, 2017-07, Vol.102 (7), p.638-638</ispartof><rights>Published by the BMJ Publishing Group Limited. For permission to use (where not already granted under a licence) please go to http://www.bmj.com/company/products-services/rights-and-licensing</rights><rights>Published by the BMJ Publishing Group Limited. For permission to use (where not already granted under a licence) please go to http://www.bmj.com/company/products-services/rights-and-licensing/.</rights><rights>Copyright: 2017 Published by the BMJ Publishing Group Limited. For permission to use (where not already granted under a licence) please go to http://www.bmj.com/company/products-services/rights-and-licensing</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-b458t-8b210f79d02a24a4c11e45237c1c73ba9beab5646d94c669f5f5c6fb3010e75d3</citedby><cites>FETCH-LOGICAL-b458t-8b210f79d02a24a4c11e45237c1c73ba9beab5646d94c669f5f5c6fb3010e75d3</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://www.proquest.com/docview/1912062431/fulltextPDF?pq-origsite=primo$$EPDF$$P50$$Gproquest$$H</linktopdf><linktohtml>$$Uhttps://www.proquest.com/docview/1912062431?pq-origsite=primo$$EHTML$$P50$$Gproquest$$H</linktohtml><link.rule.ids>314,780,784,21378,21394,27924,27925,33611,33612,33877,33878,43733,43880,74093,74269</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/28130217$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Khayat, Abdullah</creatorcontrib><creatorcontrib>Narang, Indra</creatorcontrib><creatorcontrib>Bin-Hasan, Saadoun</creatorcontrib><creatorcontrib>Amin, Reshma</creatorcontrib><creatorcontrib>Al-Saleh, Suhail</creatorcontrib><title>Longitudinal evaluation of sleep disordered breathing in infants with Prader-Willi syndrome</title><title>Archives of disease in childhood</title><addtitle>Arch Dis Child</addtitle><description>ObjectiveTo evaluate the course of sleep disordered breathing (SDB) in infants with Prader-Willi syndrome (PWS).DesignRetrospective longitudinal observational study.SettingSleep laboratory at The Hospital for Sick Children, Toronto, Canada.PatientsInfants with PWS.Main outcome measuresThe natural history of SDB in infants with PWS within 2 years from baseline assessment.ResultsWe identified 28 (12 male) infants with PWS who had a baseline polysomnography (PSG) at a median age (interquartile (IQR)) of 0.9 (0.5, 1.1) years. The median central apnoea index (CAI) at baseline was 6.6 events/hour (IQR 2.6, 12.1). Of these, 15/28 (53%) infants with PWS were diagnosed with significant central sleep apnoea (CSA) (CAI≥5 events/hour). Median age (IQR) at follow-up PSG was 2.1 (1.5, 2.6) years. The median CAI improved from 6.6 to 2.3 events/hour (p<0.0001). Only four infants with PWS had persistent CSA at the time of the follow-up PSG. Furthermore, three infants with PWS were diagnosed with mild-to-moderate obstructive sleep apnoea (OSA) that has improved at follow-up studies whereas two patients with PWS with no evidence of OSA at baseline were diagnosed with severe OSA on the follow-up PSG requiring adenotonsillectomy. The overall median obstructive apnoea–hypopnoea index was similar between baseline and follow-up studies (0.6 and 0.8, respectively, p=0.91).ConclusionsCSA is prevalent in infants with PWS but usually improves with age. However, these patients continue to require ongoing PSG surveillance because some infants will have persistent CSA and others are at risk of developing OSA.</description><subject>Age</subject><subject>Apnea</subject><subject>Care and treatment</subject><subject>Child, Preschool</subject><subject>Children</subject><subject>Endocrine therapy</subject><subject>Female</subject><subject>Genetic Disorders</subject><subject>Growth disorders</subject><subject>Growth hormones</subject><subject>Health aspects</subject><subject>Hospitals</subject><subject>Humans</subject><subject>Infant</subject><subject>Infants</subject><subject>Longitudinal Studies</subject><subject>Male</subject><subject>Observational studies</subject><subject>Oxygen therapy</subject><subject>Pediatrics</subject><subject>Polysomnography</subject><subject>Prader-Willi syndrome</subject><subject>Prader-Willi Syndrome - complications</subject><subject>Prospective Studies</subject><subject>Respiration</subject><subject>Sleep</subject><subject>Sleep apnea</subject><subject>Sleep Apnea Syndromes - etiology</subject><subject>Sleep Apnea, Central - etiology</subject><subject>Sleep deprivation</subject><subject>Sleep disorders</subject><subject>Young Children</subject><issn>0003-9888</issn><issn>1468-2044</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2017</creationdate><recordtype>article</recordtype><sourceid>ALSLI</sourceid><sourceid>CJNVE</sourceid><sourceid>M0P</sourceid><recordid>eNqNkU2LFDEQhhtR3HH1L0jAi5fWfHU6OS6DusLAelA8eAjppHomQzoZk-7V_feb3V5FvCgEAuGpN1X1NA0i-A0hTLw12R6cL_bgg2spJqJlhFAlHzUbwoWsT5w_bjYYY9YqKeVZ86yUI8aESsmeNmdUEoYp6TfNt12Kez8vzkcTEFybsJjZp4jSiEoAOKH6T8oOMjg0ZDDzwcc98rGe0cS5oB9-PqBP2VSk_epD8KjcRJfTBM-bJ6MJBV483OfNl_fvPm8v293Vh4_bi1078E7OrRwowWOvHKaGcsMtIcA7ynpLbM8GowYwQye4cIpbIdTYjZ0V48AwwdB3jp03r9fcU07fFyiznupuIAQTIS1FE0UE6xVm8t-oFLQXuOtURV_9hR7TkuuW7gMpFpQzUql2pfYmgPbRpjjDz9mmEGAPus65vdIXHRaKqP4-Va68zamUDKM-ZT-ZfKMJ1ndu9Z9u9Z1bvbqtpS8fGlqGCdzvwl8yK8BWYJiO_x97C_Khsvo</recordid><startdate>20170701</startdate><enddate>20170701</enddate><creator>Khayat, Abdullah</creator><creator>Narang, Indra</creator><creator>Bin-Hasan, Saadoun</creator><creator>Amin, Reshma</creator><creator>Al-Saleh, Suhail</creator><general>BMJ Publishing Group Ltd</general><general>BMJ Publishing Group LTD</general><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>0-V</scope><scope>3V.</scope><scope>7X7</scope><scope>7XB</scope><scope>88B</scope><scope>88E</scope><scope>88I</scope><scope>8A4</scope><scope>8AF</scope><scope>8FE</scope><scope>8FH</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>ABUWG</scope><scope>AEUYN</scope><scope>AFKRA</scope><scope>ALSLI</scope><scope>AN0</scope><scope>AZQEC</scope><scope>BBNVY</scope><scope>BENPR</scope><scope>BHPHI</scope><scope>BTHHO</scope><scope>CCPQU</scope><scope>CJNVE</scope><scope>DWQXO</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>GNUQQ</scope><scope>HCIFZ</scope><scope>K9-</scope><scope>K9.</scope><scope>LK8</scope><scope>M0P</scope><scope>M0R</scope><scope>M0S</scope><scope>M1P</scope><scope>M2P</scope><scope>M7P</scope><scope>PQEDU</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>Q9U</scope><scope>7X8</scope><scope>7T5</scope><scope>H94</scope></search><sort><creationdate>20170701</creationdate><title>Longitudinal evaluation of sleep disordered breathing in infants with Prader-Willi syndrome</title><author>Khayat, Abdullah ; Narang, Indra ; Bin-Hasan, Saadoun ; Amin, Reshma ; Al-Saleh, Suhail</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-b458t-8b210f79d02a24a4c11e45237c1c73ba9beab5646d94c669f5f5c6fb3010e75d3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2017</creationdate><topic>Age</topic><topic>Apnea</topic><topic>Care and treatment</topic><topic>Child, Preschool</topic><topic>Children</topic><topic>Endocrine therapy</topic><topic>Female</topic><topic>Genetic Disorders</topic><topic>Growth disorders</topic><topic>Growth hormones</topic><topic>Health aspects</topic><topic>Hospitals</topic><topic>Humans</topic><topic>Infant</topic><topic>Infants</topic><topic>Longitudinal Studies</topic><topic>Male</topic><topic>Observational studies</topic><topic>Oxygen therapy</topic><topic>Pediatrics</topic><topic>Polysomnography</topic><topic>Prader-Willi syndrome</topic><topic>Prader-Willi Syndrome - complications</topic><topic>Prospective Studies</topic><topic>Respiration</topic><topic>Sleep</topic><topic>Sleep apnea</topic><topic>Sleep Apnea Syndromes - etiology</topic><topic>Sleep Apnea, Central - etiology</topic><topic>Sleep deprivation</topic><topic>Sleep disorders</topic><topic>Young Children</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Khayat, Abdullah</creatorcontrib><creatorcontrib>Narang, Indra</creatorcontrib><creatorcontrib>Bin-Hasan, Saadoun</creatorcontrib><creatorcontrib>Amin, Reshma</creatorcontrib><creatorcontrib>Al-Saleh, Suhail</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>ProQuest Social Sciences Premium Collection【Remote access available】</collection><collection>ProQuest Central (Corporate)</collection><collection>Health & Medical Collection (Proquest)</collection><collection>ProQuest Central (purchase pre-March 2016)</collection><collection>Education Database (Alumni Edition)</collection><collection>Medical Database (Alumni Edition)</collection><collection>Science Database (Alumni Edition)</collection><collection>Education Periodicals</collection><collection>STEM Database</collection><collection>ProQuest SciTech Collection</collection><collection>ProQuest Natural Science Collection</collection><collection>Hospital Premium Collection</collection><collection>Hospital Premium Collection (Alumni Edition)</collection><collection>ProQuest Central (Alumni) (purchase pre-March 2016)</collection><collection>ProQuest Central (Alumni)</collection><collection>ProQuest One Sustainability</collection><collection>ProQuest Central</collection><collection>Social Science Premium Collection (Proquest) (PQ_SDU_P3)</collection><collection>British Nursing Database</collection><collection>ProQuest Central Essentials</collection><collection>Biological Science Collection</collection><collection>ProQuest Central</collection><collection>ProQuest Natural Science Collection</collection><collection>BMJ Journals</collection><collection>ProQuest One Community College</collection><collection>Education Collection (Proquest) (PQ_SDU_P3)</collection><collection>ProQuest Central</collection><collection>Health Research Premium Collection</collection><collection>Health Research Premium Collection (Alumni)</collection><collection>ProQuest Central Student</collection><collection>SciTech Premium Collection (Proquest) (PQ_SDU_P3)</collection><collection>Consumer Health Database (Alumni Edition)</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>Biological Sciences</collection><collection>Education Database (ProQuest)</collection><collection>Family Health Database (Proquest)</collection><collection>Health & Medical Collection (Alumni Edition)</collection><collection>PML(ProQuest Medical Library)</collection><collection>Science Database (ProQuest)</collection><collection>Biological Science Database</collection><collection>ProQuest One Education</collection><collection>ProQuest One Academic Eastern Edition (DO NOT USE)</collection><collection>ProQuest One Academic</collection><collection>ProQuest One Academic UKI Edition</collection><collection>ProQuest Central Basic</collection><collection>MEDLINE - Academic</collection><collection>Immunology Abstracts</collection><collection>AIDS and Cancer Research Abstracts</collection><jtitle>Archives of disease in childhood</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Khayat, Abdullah</au><au>Narang, Indra</au><au>Bin-Hasan, Saadoun</au><au>Amin, Reshma</au><au>Al-Saleh, Suhail</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Longitudinal evaluation of sleep disordered breathing in infants with Prader-Willi syndrome</atitle><jtitle>Archives of disease in childhood</jtitle><addtitle>Arch Dis Child</addtitle><date>2017-07-01</date><risdate>2017</risdate><volume>102</volume><issue>7</issue><spage>638</spage><epage>638</epage><pages>638-638</pages><issn>0003-9888</issn><eissn>1468-2044</eissn><abstract>ObjectiveTo evaluate the course of sleep disordered breathing (SDB) in infants with Prader-Willi syndrome (PWS).DesignRetrospective longitudinal observational study.SettingSleep laboratory at The Hospital for Sick Children, Toronto, Canada.PatientsInfants with PWS.Main outcome measuresThe natural history of SDB in infants with PWS within 2 years from baseline assessment.ResultsWe identified 28 (12 male) infants with PWS who had a baseline polysomnography (PSG) at a median age (interquartile (IQR)) of 0.9 (0.5, 1.1) years. The median central apnoea index (CAI) at baseline was 6.6 events/hour (IQR 2.6, 12.1). Of these, 15/28 (53%) infants with PWS were diagnosed with significant central sleep apnoea (CSA) (CAI≥5 events/hour). Median age (IQR) at follow-up PSG was 2.1 (1.5, 2.6) years. The median CAI improved from 6.6 to 2.3 events/hour (p<0.0001). Only four infants with PWS had persistent CSA at the time of the follow-up PSG. Furthermore, three infants with PWS were diagnosed with mild-to-moderate obstructive sleep apnoea (OSA) that has improved at follow-up studies whereas two patients with PWS with no evidence of OSA at baseline were diagnosed with severe OSA on the follow-up PSG requiring adenotonsillectomy. The overall median obstructive apnoea–hypopnoea index was similar between baseline and follow-up studies (0.6 and 0.8, respectively, p=0.91).ConclusionsCSA is prevalent in infants with PWS but usually improves with age. However, these patients continue to require ongoing PSG surveillance because some infants will have persistent CSA and others are at risk of developing OSA.</abstract><cop>England</cop><pub>BMJ Publishing Group Ltd</pub><pmid>28130217</pmid><doi>10.1136/archdischild-2016-311298</doi><tpages>5</tpages></addata></record> |
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| ispartof | Archives of disease in childhood, 2017-07, Vol.102 (7), p.638-638 |
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| subjects | Age Apnea Care and treatment Child, Preschool Children Endocrine therapy Female Genetic Disorders Growth disorders Growth hormones Health aspects Hospitals Humans Infant Infants Longitudinal Studies Male Observational studies Oxygen therapy Pediatrics Polysomnography Prader-Willi syndrome Prader-Willi Syndrome - complications Prospective Studies Respiration Sleep Sleep apnea Sleep Apnea Syndromes - etiology Sleep Apnea, Central - etiology Sleep deprivation Sleep disorders Young Children |
| title | Longitudinal evaluation of sleep disordered breathing in infants with Prader-Willi syndrome |
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