Magnetic Resonance Imaging Detects Progression of Lung Disease and Impact of Newborn Screening in Preschool Children with Cystic Fibrosis
Previous cross-sectional studies have demonstrated that chest magnetic resonance imaging (MRI) is sensitive to detect early lung disease in infants and preschool children with cystic fibrosis (CF) without radiation exposure. However, the ability of MRI to detect the progression of lung disease and t...
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| Veröffentlicht in: | American journal of respiratory and critical care medicine 2021-10, Vol.204 (8), p.943-953 |
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| creator | Stahl, Mirjam Steinke, Eva Graeber, Simon Y Joachim, Cornelia Seitz, Christoph Kauczor, Hans-Ulrich Eichinger, Monika Hämmerling, Susanne Sommerburg, Olaf Wielpütz, Mark O Mall, Marcus A |
| description | Previous cross-sectional studies have demonstrated that chest magnetic resonance imaging (MRI) is sensitive to detect early lung disease in infants and preschool children with cystic fibrosis (CF) without radiation exposure. However, the ability of MRI to detect the progression of lung disease and the impact of early diagnosis in preschool children with CF remains unknown.
To investigate the potential of MRI to detect progression of early lung disease and impact of early diagnosis by CF newborn screening (NBS) in preschool children with CF.
An annual MRI was performed from diagnosis over 4 years in a cohort of 96 preschool children with CF (age, 0-4 yr) who received concurrent diagnoses on the basis of NBS (
= 28) or clinical symptoms (
= 68). MRI scans were evaluated using a dedicated morphofunctional score, and the relationship between longitudinal MRI score and respiratory symptoms, pulmonary exacerbations, upper airway microbiology, and mode of diagnosis was determined.
The MRI global score increased in the total cohort of children with CF during preschool years (
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| doi_str_mv | 10.1164/rccm.202102-0278OC |
| format | Article |
| fullrecord | <record><control><sourceid>proquest_cross</sourceid><recordid>TN_cdi_proquest_journals_2586553403</recordid><sourceformat>XML</sourceformat><sourcesystem>PC</sourcesystem><sourcerecordid>2586553403</sourcerecordid><originalsourceid>FETCH-LOGICAL-c331t-190762db46dfe8d97e0a30c300561883e0e15bf28bcfb7c3d4fd1ad8f67cfd673</originalsourceid><addsrcrecordid>eNo9kNtqGzEQhkVpaZw0L9CLIuj1pqPDruTLss0JnKTkALlbtNLIlrElV1oT8gh96-zitFczMP9h-Aj5yuCMsUb-yNZuzzhwBrwCrvRd-4HMWC3qSs4VfBx3UKKScv58RI5LWQMwrhl8JkdCci0UyBn5e2OWEYdg6T2WFE20SK-3Zhnikv7CAe1Q6O-clhlLCSnS5OliP91CQVOQmuhG_c7YYTrd4kufcqQPNiPGKSPE0Y7FrlLa0HYVNi5jpC9hWNH2tUy9F6HPqYTyhXzyZlPw9H2ekKeL88f2qlrcXV63PxeVFYINFZuDarjrZeM8ajdXCEaAFQB1w7QWCMjq3nPdW98rK5z0jhmnfaOsd40SJ-T7IXeX0589lqFbp32OY2XHa93UtZAgRhU_qOz4XMnou10OW5NfOwbdRL-b6HcH-t2B_mj69h6977fo_lv-4RZvrXCDGw</addsrcrecordid><sourcetype>Aggregation Database</sourcetype><iscdi>true</iscdi><recordtype>article</recordtype><pqid>2586553403</pqid></control><display><type>article</type><title>Magnetic Resonance Imaging Detects Progression of Lung Disease and Impact of Newborn Screening in Preschool Children with Cystic Fibrosis</title><source>MEDLINE</source><source>American Thoracic Society (ATS) Journals Online</source><source>EZB-FREE-00999 freely available EZB journals</source><source>Alma/SFX Local Collection</source><creator>Stahl, Mirjam ; Steinke, Eva ; Graeber, Simon Y ; Joachim, Cornelia ; Seitz, Christoph ; Kauczor, Hans-Ulrich ; Eichinger, Monika ; Hämmerling, Susanne ; Sommerburg, Olaf ; Wielpütz, Mark O ; Mall, Marcus A</creator><creatorcontrib>Stahl, Mirjam ; Steinke, Eva ; Graeber, Simon Y ; Joachim, Cornelia ; Seitz, Christoph ; Kauczor, Hans-Ulrich ; Eichinger, Monika ; Hämmerling, Susanne ; Sommerburg, Olaf ; Wielpütz, Mark O ; Mall, Marcus A</creatorcontrib><description>Previous cross-sectional studies have demonstrated that chest magnetic resonance imaging (MRI) is sensitive to detect early lung disease in infants and preschool children with cystic fibrosis (CF) without radiation exposure. However, the ability of MRI to detect the progression of lung disease and the impact of early diagnosis in preschool children with CF remains unknown.
To investigate the potential of MRI to detect progression of early lung disease and impact of early diagnosis by CF newborn screening (NBS) in preschool children with CF.
An annual MRI was performed from diagnosis over 4 years in a cohort of 96 preschool children with CF (age, 0-4 yr) who received concurrent diagnoses on the basis of NBS (
= 28) or clinical symptoms (
= 68). MRI scans were evaluated using a dedicated morphofunctional score, and the relationship between longitudinal MRI score and respiratory symptoms, pulmonary exacerbations, upper airway microbiology, and mode of diagnosis was determined.
The MRI global score increased in the total cohort of children with CF during preschool years (
< 0.001) and was associated with cough, pulmonary exacerbations (
< 0.0001), and the detection of
and
(
< 0.05). MRI-defined abnormalities in lung morphology-especially airway wall thickening/bronchiectasis-were lower in children with CF diagnosed by NBS than in children with clinically diagnosed CF throughout the observation period (
< 0.01).
MRI detected progression of early lung disease and benefits of early diagnosis by NBS in preschool children with CF. These findings support MRI as a sensitive outcome measure for diagnostic monitoring and early intervention trials in preschool children with CF. Clinical trial registered with www.clinicaltrials.gov (NCT02270476).</description><identifier>ISSN: 1073-449X</identifier><identifier>EISSN: 1535-4970</identifier><identifier>DOI: 10.1164/rccm.202102-0278OC</identifier><identifier>PMID: 34283704</identifier><language>eng</language><publisher>United States: American Thoracic Society</publisher><subject>Child, Preschool ; Childrens health ; Cystic fibrosis ; Cystic Fibrosis - diagnostic imaging ; Cystic Fibrosis - physiopathology ; Disease Progression ; Early Diagnosis ; Female ; Humans ; Infant ; Infant, Newborn ; Linear Models ; Longitudinal Studies ; Magnetic Resonance Imaging ; Male ; Medical screening ; Neonatal Screening ; Newborn babies ; Preschool children ; Prospective Studies</subject><ispartof>American journal of respiratory and critical care medicine, 2021-10, Vol.204 (8), p.943-953</ispartof><rights>Copyright American Thoracic Society Oct 15, 2021</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c331t-190762db46dfe8d97e0a30c300561883e0e15bf28bcfb7c3d4fd1ad8f67cfd673</citedby><cites>FETCH-LOGICAL-c331t-190762db46dfe8d97e0a30c300561883e0e15bf28bcfb7c3d4fd1ad8f67cfd673</cites><orcidid>0000-0002-4057-2199 ; 0000-0001-6962-037X ; 0000-0001-6941-8315 ; 0000-0002-1666-8924</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>314,780,784,4025,27924,27925</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/34283704$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Stahl, Mirjam</creatorcontrib><creatorcontrib>Steinke, Eva</creatorcontrib><creatorcontrib>Graeber, Simon Y</creatorcontrib><creatorcontrib>Joachim, Cornelia</creatorcontrib><creatorcontrib>Seitz, Christoph</creatorcontrib><creatorcontrib>Kauczor, Hans-Ulrich</creatorcontrib><creatorcontrib>Eichinger, Monika</creatorcontrib><creatorcontrib>Hämmerling, Susanne</creatorcontrib><creatorcontrib>Sommerburg, Olaf</creatorcontrib><creatorcontrib>Wielpütz, Mark O</creatorcontrib><creatorcontrib>Mall, Marcus A</creatorcontrib><title>Magnetic Resonance Imaging Detects Progression of Lung Disease and Impact of Newborn Screening in Preschool Children with Cystic Fibrosis</title><title>American journal of respiratory and critical care medicine</title><addtitle>Am J Respir Crit Care Med</addtitle><description>Previous cross-sectional studies have demonstrated that chest magnetic resonance imaging (MRI) is sensitive to detect early lung disease in infants and preschool children with cystic fibrosis (CF) without radiation exposure. However, the ability of MRI to detect the progression of lung disease and the impact of early diagnosis in preschool children with CF remains unknown.
To investigate the potential of MRI to detect progression of early lung disease and impact of early diagnosis by CF newborn screening (NBS) in preschool children with CF.
An annual MRI was performed from diagnosis over 4 years in a cohort of 96 preschool children with CF (age, 0-4 yr) who received concurrent diagnoses on the basis of NBS (
= 28) or clinical symptoms (
= 68). MRI scans were evaluated using a dedicated morphofunctional score, and the relationship between longitudinal MRI score and respiratory symptoms, pulmonary exacerbations, upper airway microbiology, and mode of diagnosis was determined.
The MRI global score increased in the total cohort of children with CF during preschool years (
< 0.001) and was associated with cough, pulmonary exacerbations (
< 0.0001), and the detection of
and
(
< 0.05). MRI-defined abnormalities in lung morphology-especially airway wall thickening/bronchiectasis-were lower in children with CF diagnosed by NBS than in children with clinically diagnosed CF throughout the observation period (
< 0.01).
MRI detected progression of early lung disease and benefits of early diagnosis by NBS in preschool children with CF. These findings support MRI as a sensitive outcome measure for diagnostic monitoring and early intervention trials in preschool children with CF. Clinical trial registered with www.clinicaltrials.gov (NCT02270476).</description><subject>Child, Preschool</subject><subject>Childrens health</subject><subject>Cystic fibrosis</subject><subject>Cystic Fibrosis - diagnostic imaging</subject><subject>Cystic Fibrosis - physiopathology</subject><subject>Disease Progression</subject><subject>Early Diagnosis</subject><subject>Female</subject><subject>Humans</subject><subject>Infant</subject><subject>Infant, Newborn</subject><subject>Linear Models</subject><subject>Longitudinal Studies</subject><subject>Magnetic Resonance Imaging</subject><subject>Male</subject><subject>Medical screening</subject><subject>Neonatal Screening</subject><subject>Newborn babies</subject><subject>Preschool children</subject><subject>Prospective Studies</subject><issn>1073-449X</issn><issn>1535-4970</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2021</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNo9kNtqGzEQhkVpaZw0L9CLIuj1pqPDruTLss0JnKTkALlbtNLIlrElV1oT8gh96-zitFczMP9h-Aj5yuCMsUb-yNZuzzhwBrwCrvRd-4HMWC3qSs4VfBx3UKKScv58RI5LWQMwrhl8JkdCci0UyBn5e2OWEYdg6T2WFE20SK-3Zhnikv7CAe1Q6O-clhlLCSnS5OliP91CQVOQmuhG_c7YYTrd4kufcqQPNiPGKSPE0Y7FrlLa0HYVNi5jpC9hWNH2tUy9F6HPqYTyhXzyZlPw9H2ekKeL88f2qlrcXV63PxeVFYINFZuDarjrZeM8ajdXCEaAFQB1w7QWCMjq3nPdW98rK5z0jhmnfaOsd40SJ-T7IXeX0589lqFbp32OY2XHa93UtZAgRhU_qOz4XMnou10OW5NfOwbdRL-b6HcH-t2B_mj69h6977fo_lv-4RZvrXCDGw</recordid><startdate>20211015</startdate><enddate>20211015</enddate><creator>Stahl, Mirjam</creator><creator>Steinke, Eva</creator><creator>Graeber, Simon Y</creator><creator>Joachim, Cornelia</creator><creator>Seitz, Christoph</creator><creator>Kauczor, Hans-Ulrich</creator><creator>Eichinger, Monika</creator><creator>Hämmerling, Susanne</creator><creator>Sommerburg, Olaf</creator><creator>Wielpütz, Mark O</creator><creator>Mall, Marcus A</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><orcidid>https://orcid.org/0000-0002-4057-2199</orcidid><orcidid>https://orcid.org/0000-0001-6962-037X</orcidid><orcidid>https://orcid.org/0000-0001-6941-8315</orcidid><orcidid>https://orcid.org/0000-0002-1666-8924</orcidid></search><sort><creationdate>20211015</creationdate><title>Magnetic Resonance Imaging Detects Progression of Lung Disease and Impact of Newborn Screening in Preschool Children with Cystic Fibrosis</title><author>Stahl, Mirjam ; Steinke, Eva ; Graeber, Simon Y ; Joachim, Cornelia ; Seitz, Christoph ; Kauczor, Hans-Ulrich ; Eichinger, Monika ; Hämmerling, Susanne ; Sommerburg, Olaf ; Wielpütz, Mark O ; Mall, Marcus A</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c331t-190762db46dfe8d97e0a30c300561883e0e15bf28bcfb7c3d4fd1ad8f67cfd673</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2021</creationdate><topic>Child, Preschool</topic><topic>Childrens health</topic><topic>Cystic fibrosis</topic><topic>Cystic Fibrosis - diagnostic imaging</topic><topic>Cystic Fibrosis - physiopathology</topic><topic>Disease Progression</topic><topic>Early Diagnosis</topic><topic>Female</topic><topic>Humans</topic><topic>Infant</topic><topic>Infant, Newborn</topic><topic>Linear Models</topic><topic>Longitudinal Studies</topic><topic>Magnetic Resonance Imaging</topic><topic>Male</topic><topic>Medical screening</topic><topic>Neonatal Screening</topic><topic>Newborn babies</topic><topic>Preschool children</topic><topic>Prospective Studies</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Stahl, Mirjam</creatorcontrib><creatorcontrib>Steinke, Eva</creatorcontrib><creatorcontrib>Graeber, Simon Y</creatorcontrib><creatorcontrib>Joachim, Cornelia</creatorcontrib><creatorcontrib>Seitz, Christoph</creatorcontrib><creatorcontrib>Kauczor, Hans-Ulrich</creatorcontrib><creatorcontrib>Eichinger, Monika</creatorcontrib><creatorcontrib>Hämmerling, Susanne</creatorcontrib><creatorcontrib>Sommerburg, Olaf</creatorcontrib><creatorcontrib>Wielpütz, Mark O</creatorcontrib><creatorcontrib>Mall, Marcus A</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><jtitle>American journal of respiratory and critical care medicine</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Stahl, Mirjam</au><au>Steinke, Eva</au><au>Graeber, Simon Y</au><au>Joachim, Cornelia</au><au>Seitz, Christoph</au><au>Kauczor, Hans-Ulrich</au><au>Eichinger, Monika</au><au>Hämmerling, Susanne</au><au>Sommerburg, Olaf</au><au>Wielpütz, Mark O</au><au>Mall, Marcus A</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Magnetic Resonance Imaging Detects Progression of Lung Disease and Impact of Newborn Screening in Preschool Children with Cystic Fibrosis</atitle><jtitle>American journal of respiratory and critical care medicine</jtitle><addtitle>Am J Respir Crit Care Med</addtitle><date>2021-10-15</date><risdate>2021</risdate><volume>204</volume><issue>8</issue><spage>943</spage><epage>953</epage><pages>943-953</pages><issn>1073-449X</issn><eissn>1535-4970</eissn><abstract>Previous cross-sectional studies have demonstrated that chest magnetic resonance imaging (MRI) is sensitive to detect early lung disease in infants and preschool children with cystic fibrosis (CF) without radiation exposure. However, the ability of MRI to detect the progression of lung disease and the impact of early diagnosis in preschool children with CF remains unknown.
To investigate the potential of MRI to detect progression of early lung disease and impact of early diagnosis by CF newborn screening (NBS) in preschool children with CF.
An annual MRI was performed from diagnosis over 4 years in a cohort of 96 preschool children with CF (age, 0-4 yr) who received concurrent diagnoses on the basis of NBS (
= 28) or clinical symptoms (
= 68). MRI scans were evaluated using a dedicated morphofunctional score, and the relationship between longitudinal MRI score and respiratory symptoms, pulmonary exacerbations, upper airway microbiology, and mode of diagnosis was determined.
The MRI global score increased in the total cohort of children with CF during preschool years (
< 0.001) and was associated with cough, pulmonary exacerbations (
< 0.0001), and the detection of
and
(
< 0.05). MRI-defined abnormalities in lung morphology-especially airway wall thickening/bronchiectasis-were lower in children with CF diagnosed by NBS than in children with clinically diagnosed CF throughout the observation period (
< 0.01).
MRI detected progression of early lung disease and benefits of early diagnosis by NBS in preschool children with CF. These findings support MRI as a sensitive outcome measure for diagnostic monitoring and early intervention trials in preschool children with CF. Clinical trial registered with www.clinicaltrials.gov (NCT02270476).</abstract><cop>United States</cop><pub>American Thoracic Society</pub><pmid>34283704</pmid><doi>10.1164/rccm.202102-0278OC</doi><tpages>11</tpages><orcidid>https://orcid.org/0000-0002-4057-2199</orcidid><orcidid>https://orcid.org/0000-0001-6962-037X</orcidid><orcidid>https://orcid.org/0000-0001-6941-8315</orcidid><orcidid>https://orcid.org/0000-0002-1666-8924</orcidid></addata></record> |
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| identifier | ISSN: 1073-449X |
| ispartof | American journal of respiratory and critical care medicine, 2021-10, Vol.204 (8), p.943-953 |
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| language | eng |
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| source | MEDLINE; American Thoracic Society (ATS) Journals Online; EZB-FREE-00999 freely available EZB journals; Alma/SFX Local Collection |
| subjects | Child, Preschool Childrens health Cystic fibrosis Cystic Fibrosis - diagnostic imaging Cystic Fibrosis - physiopathology Disease Progression Early Diagnosis Female Humans Infant Infant, Newborn Linear Models Longitudinal Studies Magnetic Resonance Imaging Male Medical screening Neonatal Screening Newborn babies Preschool children Prospective Studies |
| title | Magnetic Resonance Imaging Detects Progression of Lung Disease and Impact of Newborn Screening in Preschool Children with Cystic Fibrosis |
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