Association of Neonatal Hospital Length of Stay with Lung Function in Primary Ciliary Dyskinesia
Primary ciliary dyskinesia (PCD), an inherited lung disease, is characterized by abnormal ciliary function leading to progressive bronchiectasis. There is wide variability in respiratory disease severity at birth and later in life. To evaluate the association between neonatal hospital length of stay...
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| Veröffentlicht in: | Annals of the American Thoracic Society 2022-11, Vol.19 (11), p.1865-1870 |
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| creator | Wee, Wallace B Leigh, Margaret W Davis, Stephanie D Rosenfeld, Margaret Sullivan, Kelli M Sawras, Michael G Ferkol, Thomas W Knowles, Michael R Milla, Carlos Sagel, Scott D Zariwala, Maimoona A Pullenayegum, Eleanor Dell, Sharon D |
| description | Primary ciliary dyskinesia (PCD), an inherited lung disease, is characterized by abnormal ciliary function leading to progressive bronchiectasis. There is wide variability in respiratory disease severity at birth and later in life.
To evaluate the association between neonatal hospital length of stay (neonatal-LOS) and supplemental oxygen duration (SuppO
) with lung function in pediatric PCD. We hypothesized that longer neonatal-LOS and SuppO
are associated with worse lung function (i.e., forced expiratory volume in 1 second percent predicted [FEV
pp]).
We performed a secondary analysis of the Genetic Disorders of Mucociliary Clearance Consortium prospective longitudinal multicenter cohort study. Participants enrolled, during 2006-2011, were |
| doi_str_mv | 10.1513/AnnalsATS.202202-116OC |
| format | Article |
| fullrecord | <record><control><sourceid>proquest_pubme</sourceid><recordid>TN_cdi_pubmedcentral_primary_oai_pubmedcentral_nih_gov_9667809</recordid><sourceformat>XML</sourceformat><sourcesystem>PC</sourcesystem><sourcerecordid>2673358605</sourcerecordid><originalsourceid>FETCH-LOGICAL-c394t-1dddc97558e44cf7e556cbeef129b327bb6295e894f853e4fd6920ffffc215a83</originalsourceid><addsrcrecordid>eNpdkVFLwzAQx4MoKrqvIAVffOlskiZpXoQxnQpDhc3nmKbpjHbJbFpl3950nUM9Anfh_v_jjh8AZzAZQgLx5chaWfnRfDZECQovhpA-jvfAMcKIxJQiuL-peUw5xkdg4P1bEiIjMGP8EBxhQgljmB6Dl5H3ThnZGGcjV0YP2lnZyCq6c35lumKq7aJ57XqzRq6jLxM-09Yuoklr1cZmbPRUm6Ws19HYVKbL12v_bqz2Rp6CgzIsqwfbfAKeJzfz8V08fby9H4-mscI8bWJYFIXijJBMp6kqmSaEqlzrEiKeY8TynCJOdMbTMiNYp2VBOUrKEApBIjN8Aq76uas2X-pCadvUshKrfjHhpBF_O9a8ioX7FJxSliU8DLjYDqjdR6t9I5bGK11V0mrXeoEow5hkNCFBev5P-ubaumMiEMMM0o5DUNFepWrnfa3L3TIwER1HseMoeo5iwzEYz36fsrP9UMPfAuac0A</addsrcrecordid><sourcetype>Open Access Repository</sourcetype><iscdi>true</iscdi><recordtype>article</recordtype><pqid>2737162325</pqid></control><display><type>article</type><title>Association of Neonatal Hospital Length of Stay with Lung Function in Primary Ciliary Dyskinesia</title><source>MEDLINE</source><source>American Thoracic Society Journals</source><source>Alma/SFX Local Collection</source><creator>Wee, Wallace B ; Leigh, Margaret W ; Davis, Stephanie D ; Rosenfeld, Margaret ; Sullivan, Kelli M ; Sawras, Michael G ; Ferkol, Thomas W ; Knowles, Michael R ; Milla, Carlos ; Sagel, Scott D ; Zariwala, Maimoona A ; Pullenayegum, Eleanor ; Dell, Sharon D</creator><creatorcontrib>Wee, Wallace B ; Leigh, Margaret W ; Davis, Stephanie D ; Rosenfeld, Margaret ; Sullivan, Kelli M ; Sawras, Michael G ; Ferkol, Thomas W ; Knowles, Michael R ; Milla, Carlos ; Sagel, Scott D ; Zariwala, Maimoona A ; Pullenayegum, Eleanor ; Dell, Sharon D</creatorcontrib><description>Primary ciliary dyskinesia (PCD), an inherited lung disease, is characterized by abnormal ciliary function leading to progressive bronchiectasis. There is wide variability in respiratory disease severity at birth and later in life.
To evaluate the association between neonatal hospital length of stay (neonatal-LOS) and supplemental oxygen duration (SuppO
) with lung function in pediatric PCD. We hypothesized that longer neonatal-LOS and SuppO
are associated with worse lung function (i.e., forced expiratory volume in 1 second percent predicted [FEV
pp]).
We performed a secondary analysis of the Genetic Disorders of Mucociliary Clearance Consortium prospective longitudinal multicenter cohort study. Participants enrolled, during 2006-2011, were <19 years old with a confirmed PCD diagnosis and followed annually for 5 years. The exposure variables were neonatal-LOS and SuppO
, counted in days since birth. The outcome, FEV
pp, was measured annually by spirometry. The associations of neonatal-LOS and SuppO
with FEV
pp were evaluated with a linear mixed-effects model with repeated measures and random intercepts, adjusted for age and ciliary ultrastructural defects.
Included were 123 participants (male, 47%; mean enrollment age, 8.3 yr [range, 0 to 18 yr]) with 578 visits (median follow-up, 5 yr). The median neonatal-LOS was 9 d (range, 1 to 90 d), and median SuppO
was 5 d (range, 0 to 180 d). Neonatal-LOS was associated with worse lung function (-0.27 FEV
pp/d [95% confidence interval, -0.53 to -0.01];
= 0.04). SuppO
was not associated with lung function.
Neonatal-LOS is associated with worse lung function in pediatric PCD, independent of age and ultrastructural defects. Future research on the mechanisms of neonatal respiratory distress and its management may help us understand the variability of lung health outcomes in PCD.</description><identifier>ISSN: 2329-6933</identifier><identifier>EISSN: 2325-6621</identifier><identifier>DOI: 10.1513/AnnalsATS.202202-116OC</identifier><identifier>PMID: 35657736</identifier><language>eng</language><publisher>United States: American Thoracic Society</publisher><subject>Adolescent ; Child ; Child, Preschool ; Ciliary Motility Disorders ; Cohort Studies ; Dyskinesia ; Hospitals ; Humans ; Infant ; Infant, Newborn ; Kartagener Syndrome - diagnosis ; Length of Stay ; Lung ; Lung diseases ; Male ; Neonatal care ; Newborn babies ; Original Research ; Pediatrics ; Prospective Studies ; Respiratory distress syndrome ; Spirometry</subject><ispartof>Annals of the American Thoracic Society, 2022-11, Vol.19 (11), p.1865-1870</ispartof><rights>Copyright American Thoracic Society Nov 2022</rights><rights>Copyright © 2022 by the American Thoracic Society 2022</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c394t-1dddc97558e44cf7e556cbeef129b327bb6295e894f853e4fd6920ffffc215a83</citedby><cites>FETCH-LOGICAL-c394t-1dddc97558e44cf7e556cbeef129b327bb6295e894f853e4fd6920ffffc215a83</cites><orcidid>0000-0002-6420-2459 ; 0000-0003-2169-9407 ; 0000-0003-1619-1393 ; 0000-0003-4291-3513 ; 0000-0001-6172-4465</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>230,314,780,784,885,27924,27925</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/35657736$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Wee, Wallace B</creatorcontrib><creatorcontrib>Leigh, Margaret W</creatorcontrib><creatorcontrib>Davis, Stephanie D</creatorcontrib><creatorcontrib>Rosenfeld, Margaret</creatorcontrib><creatorcontrib>Sullivan, Kelli M</creatorcontrib><creatorcontrib>Sawras, Michael G</creatorcontrib><creatorcontrib>Ferkol, Thomas W</creatorcontrib><creatorcontrib>Knowles, Michael R</creatorcontrib><creatorcontrib>Milla, Carlos</creatorcontrib><creatorcontrib>Sagel, Scott D</creatorcontrib><creatorcontrib>Zariwala, Maimoona A</creatorcontrib><creatorcontrib>Pullenayegum, Eleanor</creatorcontrib><creatorcontrib>Dell, Sharon D</creatorcontrib><title>Association of Neonatal Hospital Length of Stay with Lung Function in Primary Ciliary Dyskinesia</title><title>Annals of the American Thoracic Society</title><addtitle>Ann Am Thorac Soc</addtitle><description>Primary ciliary dyskinesia (PCD), an inherited lung disease, is characterized by abnormal ciliary function leading to progressive bronchiectasis. There is wide variability in respiratory disease severity at birth and later in life.
To evaluate the association between neonatal hospital length of stay (neonatal-LOS) and supplemental oxygen duration (SuppO
) with lung function in pediatric PCD. We hypothesized that longer neonatal-LOS and SuppO
are associated with worse lung function (i.e., forced expiratory volume in 1 second percent predicted [FEV
pp]).
We performed a secondary analysis of the Genetic Disorders of Mucociliary Clearance Consortium prospective longitudinal multicenter cohort study. Participants enrolled, during 2006-2011, were <19 years old with a confirmed PCD diagnosis and followed annually for 5 years. The exposure variables were neonatal-LOS and SuppO
, counted in days since birth. The outcome, FEV
pp, was measured annually by spirometry. The associations of neonatal-LOS and SuppO
with FEV
pp were evaluated with a linear mixed-effects model with repeated measures and random intercepts, adjusted for age and ciliary ultrastructural defects.
Included were 123 participants (male, 47%; mean enrollment age, 8.3 yr [range, 0 to 18 yr]) with 578 visits (median follow-up, 5 yr). The median neonatal-LOS was 9 d (range, 1 to 90 d), and median SuppO
was 5 d (range, 0 to 180 d). Neonatal-LOS was associated with worse lung function (-0.27 FEV
pp/d [95% confidence interval, -0.53 to -0.01];
= 0.04). SuppO
was not associated with lung function.
Neonatal-LOS is associated with worse lung function in pediatric PCD, independent of age and ultrastructural defects. Future research on the mechanisms of neonatal respiratory distress and its management may help us understand the variability of lung health outcomes in PCD.</description><subject>Adolescent</subject><subject>Child</subject><subject>Child, Preschool</subject><subject>Ciliary Motility Disorders</subject><subject>Cohort Studies</subject><subject>Dyskinesia</subject><subject>Hospitals</subject><subject>Humans</subject><subject>Infant</subject><subject>Infant, Newborn</subject><subject>Kartagener Syndrome - diagnosis</subject><subject>Length of Stay</subject><subject>Lung</subject><subject>Lung diseases</subject><subject>Male</subject><subject>Neonatal care</subject><subject>Newborn babies</subject><subject>Original Research</subject><subject>Pediatrics</subject><subject>Prospective Studies</subject><subject>Respiratory distress syndrome</subject><subject>Spirometry</subject><issn>2329-6933</issn><issn>2325-6621</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2022</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNpdkVFLwzAQx4MoKrqvIAVffOlskiZpXoQxnQpDhc3nmKbpjHbJbFpl3950nUM9Anfh_v_jjh8AZzAZQgLx5chaWfnRfDZECQovhpA-jvfAMcKIxJQiuL-peUw5xkdg4P1bEiIjMGP8EBxhQgljmB6Dl5H3ThnZGGcjV0YP2lnZyCq6c35lumKq7aJ57XqzRq6jLxM-09Yuoklr1cZmbPRUm6Ws19HYVKbL12v_bqz2Rp6CgzIsqwfbfAKeJzfz8V08fby9H4-mscI8bWJYFIXijJBMp6kqmSaEqlzrEiKeY8TynCJOdMbTMiNYp2VBOUrKEApBIjN8Aq76uas2X-pCadvUshKrfjHhpBF_O9a8ioX7FJxSliU8DLjYDqjdR6t9I5bGK11V0mrXeoEow5hkNCFBev5P-ubaumMiEMMM0o5DUNFepWrnfa3L3TIwER1HseMoeo5iwzEYz36fsrP9UMPfAuac0A</recordid><startdate>202211</startdate><enddate>202211</enddate><creator>Wee, Wallace B</creator><creator>Leigh, Margaret W</creator><creator>Davis, Stephanie D</creator><creator>Rosenfeld, Margaret</creator><creator>Sullivan, Kelli M</creator><creator>Sawras, Michael G</creator><creator>Ferkol, Thomas W</creator><creator>Knowles, Michael R</creator><creator>Milla, Carlos</creator><creator>Sagel, Scott D</creator><creator>Zariwala, Maimoona A</creator><creator>Pullenayegum, Eleanor</creator><creator>Dell, Sharon D</creator><general>American Thoracic Society</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>K9.</scope><scope>NAPCQ</scope><scope>7X8</scope><scope>5PM</scope><orcidid>https://orcid.org/0000-0002-6420-2459</orcidid><orcidid>https://orcid.org/0000-0003-2169-9407</orcidid><orcidid>https://orcid.org/0000-0003-1619-1393</orcidid><orcidid>https://orcid.org/0000-0003-4291-3513</orcidid><orcidid>https://orcid.org/0000-0001-6172-4465</orcidid></search><sort><creationdate>202211</creationdate><title>Association of Neonatal Hospital Length of Stay with Lung Function in Primary Ciliary Dyskinesia</title><author>Wee, Wallace B ; Leigh, Margaret W ; Davis, Stephanie D ; Rosenfeld, Margaret ; Sullivan, Kelli M ; Sawras, Michael G ; Ferkol, Thomas W ; Knowles, Michael R ; Milla, Carlos ; Sagel, Scott D ; Zariwala, Maimoona A ; Pullenayegum, Eleanor ; Dell, Sharon D</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c394t-1dddc97558e44cf7e556cbeef129b327bb6295e894f853e4fd6920ffffc215a83</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2022</creationdate><topic>Adolescent</topic><topic>Child</topic><topic>Child, Preschool</topic><topic>Ciliary Motility Disorders</topic><topic>Cohort Studies</topic><topic>Dyskinesia</topic><topic>Hospitals</topic><topic>Humans</topic><topic>Infant</topic><topic>Infant, Newborn</topic><topic>Kartagener Syndrome - diagnosis</topic><topic>Length of Stay</topic><topic>Lung</topic><topic>Lung diseases</topic><topic>Male</topic><topic>Neonatal care</topic><topic>Newborn babies</topic><topic>Original Research</topic><topic>Pediatrics</topic><topic>Prospective Studies</topic><topic>Respiratory distress syndrome</topic><topic>Spirometry</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Wee, Wallace B</creatorcontrib><creatorcontrib>Leigh, Margaret W</creatorcontrib><creatorcontrib>Davis, Stephanie D</creatorcontrib><creatorcontrib>Rosenfeld, Margaret</creatorcontrib><creatorcontrib>Sullivan, Kelli M</creatorcontrib><creatorcontrib>Sawras, Michael G</creatorcontrib><creatorcontrib>Ferkol, Thomas W</creatorcontrib><creatorcontrib>Knowles, Michael R</creatorcontrib><creatorcontrib>Milla, Carlos</creatorcontrib><creatorcontrib>Sagel, Scott D</creatorcontrib><creatorcontrib>Zariwala, Maimoona A</creatorcontrib><creatorcontrib>Pullenayegum, Eleanor</creatorcontrib><creatorcontrib>Dell, Sharon D</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 Health & Medical Complete (Alumni)</collection><collection>Nursing & Allied Health Premium</collection><collection>MEDLINE - Academic</collection><collection>PubMed Central (Full Participant titles)</collection><jtitle>Annals of the American Thoracic Society</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Wee, Wallace B</au><au>Leigh, Margaret W</au><au>Davis, Stephanie D</au><au>Rosenfeld, Margaret</au><au>Sullivan, Kelli M</au><au>Sawras, Michael G</au><au>Ferkol, Thomas W</au><au>Knowles, Michael R</au><au>Milla, Carlos</au><au>Sagel, Scott D</au><au>Zariwala, Maimoona A</au><au>Pullenayegum, Eleanor</au><au>Dell, Sharon D</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Association of Neonatal Hospital Length of Stay with Lung Function in Primary Ciliary Dyskinesia</atitle><jtitle>Annals of the American Thoracic Society</jtitle><addtitle>Ann Am Thorac Soc</addtitle><date>2022-11</date><risdate>2022</risdate><volume>19</volume><issue>11</issue><spage>1865</spage><epage>1870</epage><pages>1865-1870</pages><issn>2329-6933</issn><eissn>2325-6621</eissn><abstract>Primary ciliary dyskinesia (PCD), an inherited lung disease, is characterized by abnormal ciliary function leading to progressive bronchiectasis. There is wide variability in respiratory disease severity at birth and later in life.
To evaluate the association between neonatal hospital length of stay (neonatal-LOS) and supplemental oxygen duration (SuppO
) with lung function in pediatric PCD. We hypothesized that longer neonatal-LOS and SuppO
are associated with worse lung function (i.e., forced expiratory volume in 1 second percent predicted [FEV
pp]).
We performed a secondary analysis of the Genetic Disorders of Mucociliary Clearance Consortium prospective longitudinal multicenter cohort study. Participants enrolled, during 2006-2011, were <19 years old with a confirmed PCD diagnosis and followed annually for 5 years. The exposure variables were neonatal-LOS and SuppO
, counted in days since birth. The outcome, FEV
pp, was measured annually by spirometry. The associations of neonatal-LOS and SuppO
with FEV
pp were evaluated with a linear mixed-effects model with repeated measures and random intercepts, adjusted for age and ciliary ultrastructural defects.
Included were 123 participants (male, 47%; mean enrollment age, 8.3 yr [range, 0 to 18 yr]) with 578 visits (median follow-up, 5 yr). The median neonatal-LOS was 9 d (range, 1 to 90 d), and median SuppO
was 5 d (range, 0 to 180 d). Neonatal-LOS was associated with worse lung function (-0.27 FEV
pp/d [95% confidence interval, -0.53 to -0.01];
= 0.04). SuppO
was not associated with lung function.
Neonatal-LOS is associated with worse lung function in pediatric PCD, independent of age and ultrastructural defects. Future research on the mechanisms of neonatal respiratory distress and its management may help us understand the variability of lung health outcomes in PCD.</abstract><cop>United States</cop><pub>American Thoracic Society</pub><pmid>35657736</pmid><doi>10.1513/AnnalsATS.202202-116OC</doi><tpages>6</tpages><orcidid>https://orcid.org/0000-0002-6420-2459</orcidid><orcidid>https://orcid.org/0000-0003-2169-9407</orcidid><orcidid>https://orcid.org/0000-0003-1619-1393</orcidid><orcidid>https://orcid.org/0000-0003-4291-3513</orcidid><orcidid>https://orcid.org/0000-0001-6172-4465</orcidid></addata></record> |
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| identifier | ISSN: 2329-6933 |
| ispartof | Annals of the American Thoracic Society, 2022-11, Vol.19 (11), p.1865-1870 |
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| source | MEDLINE; American Thoracic Society Journals; Alma/SFX Local Collection |
| subjects | Adolescent Child Child, Preschool Ciliary Motility Disorders Cohort Studies Dyskinesia Hospitals Humans Infant Infant, Newborn Kartagener Syndrome - diagnosis Length of Stay Lung Lung diseases Male Neonatal care Newborn babies Original Research Pediatrics Prospective Studies Respiratory distress syndrome Spirometry |
| title | Association of Neonatal Hospital Length of Stay with Lung Function in Primary Ciliary Dyskinesia |
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