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Dysanaptic growth of lung and airway in children with post-infectious bronchiolitis obliterans

Rationale Post‐infectious bronchiolitis obliterans (PBO) is a rare form of chronic obstructive lung disease associated with small airway fibrosis following a severe insult to the lower respiratory tract. It has been suggested that PBO is a non‐progressive disease. However, evidence supporting this s...

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Published in:The clinical respiratory journal 2014-01, Vol.8 (1), p.63-71
Main Authors: Mosquera, Ricardo A., Hashmi, Syed S., Pacheco, Susan E., Reverdin, Alexandra, Chevallier, Justyna, Colasurdo, Giuseppe N.
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description Rationale Post‐infectious bronchiolitis obliterans (PBO) is a rare form of chronic obstructive lung disease associated with small airway fibrosis following a severe insult to the lower respiratory tract. It has been suggested that PBO is a non‐progressive disease. However, evidence supporting this statement is limited. In this case series, we sought to determine the changes of pulmonary function tests (PFT) over time in children with PBO. Methods Seven children with PBO, ages 6–15 years old, were retrospectively studied between 1994 and 2012. Spirometry and lung volumes tests were performed in accordance with American Thoracic Society (ATS) guidelines and were monitored over time. The average rate of change was calculated using generalized linear mixed models. Results The median baseline values for forced vital capacity (FVC), forced expiratory volume in 1 s (FEV1), the FEV1/FVC ratio and forced expiratory flow 25%–75% (FEF25%–75%) were 57%, 50%, 87% and 29%, respectively. FVC increased at a rate of 1.8% per year (P = 0.008). There was no significant change in FEV1 over time (P = 0.112). However, the FEV1/FVC ratio decreased by 2.6% per year (P 
doi_str_mv 10.1111/crj.12037
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It has been suggested that PBO is a non‐progressive disease. However, evidence supporting this statement is limited. In this case series, we sought to determine the changes of pulmonary function tests (PFT) over time in children with PBO. Methods Seven children with PBO, ages 6–15 years old, were retrospectively studied between 1994 and 2012. Spirometry and lung volumes tests were performed in accordance with American Thoracic Society (ATS) guidelines and were monitored over time. The average rate of change was calculated using generalized linear mixed models. Results The median baseline values for forced vital capacity (FVC), forced expiratory volume in 1 s (FEV1), the FEV1/FVC ratio and forced expiratory flow 25%–75% (FEF25%–75%) were 57%, 50%, 87% and 29%, respectively. FVC increased at a rate of 1.8% per year (P = 0.008). There was no significant change in FEV1 over time (P = 0.112). However, the FEV1/FVC ratio decreased by 2.6% per year (P &lt; 0.001). Conclusion PFT in childhood PBO was characterized by significant airway obstruction. Over time, FVC (lung parenchyma) increased and FEV1 (airway) remained stable, but FEV1/FVC ratio declined more than expected, suggesting a mismatch in the growth of the airway and lung parenchyma (dysanaptic growth). Further studies in larger populations are needed to validate these observations.</description><identifier>ISSN: 1752-6981</identifier><identifier>EISSN: 1752-699X</identifier><identifier>DOI: 10.1111/crj.12037</identifier><identifier>PMID: 23800208</identifier><language>eng</language><publisher>England: Blackwell Publishing Ltd</publisher><subject>Adolescent ; Airway management ; bronchiolitis obliterans ; Bronchiolitis Obliterans - diagnostic imaging ; Bronchiolitis Obliterans - etiology ; Bronchiolitis Obliterans - pathology ; Bronchiolitis Obliterans - physiopathology ; Child ; Disease Progression ; dysanaptic growth ; Female ; Forced Expiratory Volume ; Humans ; immunomodulation ; Inflammation - pathology ; Inflammation - physiopathology ; inhaled corticosteroids ; Lung - pathology ; Lung - physiopathology ; Male ; pulmonary function test ; Respiratory diseases ; Respiratory Function Tests ; Respiratory Tract Infections - complications ; Tomography, X-Ray Computed - methods ; Vital Capacity</subject><ispartof>The clinical respiratory journal, 2014-01, Vol.8 (1), p.63-71</ispartof><rights>2013 John Wiley &amp; Sons Ltd</rights><rights>2013 John Wiley &amp; Sons Ltd.</rights><rights>Copyright © 2014 John Wiley &amp; Sons Ltd</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c3917-42ab3c6d8677a8adfe10e6351e3e6192d0b3918080817f3b98dcfec6d7b1455a3</citedby><cites>FETCH-LOGICAL-c3917-42ab3c6d8677a8adfe10e6351e3e6192d0b3918080817f3b98dcfec6d7b1455a3</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://onlinelibrary.wiley.com/doi/pdf/10.1111%2Fcrj.12037$$EPDF$$P50$$Gwiley$$H</linktopdf><linktohtml>$$Uhttps://onlinelibrary.wiley.com/doi/full/10.1111%2Fcrj.12037$$EHTML$$P50$$Gwiley$$H</linktohtml><link.rule.ids>315,786,790,27957,27958,50923,51032</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/23800208$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Mosquera, Ricardo A.</creatorcontrib><creatorcontrib>Hashmi, Syed S.</creatorcontrib><creatorcontrib>Pacheco, Susan E.</creatorcontrib><creatorcontrib>Reverdin, Alexandra</creatorcontrib><creatorcontrib>Chevallier, Justyna</creatorcontrib><creatorcontrib>Colasurdo, Giuseppe N.</creatorcontrib><title>Dysanaptic growth of lung and airway in children with post-infectious bronchiolitis obliterans</title><title>The clinical respiratory journal</title><addtitle>The Clinical Respiratory Journal</addtitle><description>Rationale Post‐infectious bronchiolitis obliterans (PBO) is a rare form of chronic obstructive lung disease associated with small airway fibrosis following a severe insult to the lower respiratory tract. It has been suggested that PBO is a non‐progressive disease. However, evidence supporting this statement is limited. In this case series, we sought to determine the changes of pulmonary function tests (PFT) over time in children with PBO. Methods Seven children with PBO, ages 6–15 years old, were retrospectively studied between 1994 and 2012. Spirometry and lung volumes tests were performed in accordance with American Thoracic Society (ATS) guidelines and were monitored over time. The average rate of change was calculated using generalized linear mixed models. Results The median baseline values for forced vital capacity (FVC), forced expiratory volume in 1 s (FEV1), the FEV1/FVC ratio and forced expiratory flow 25%–75% (FEF25%–75%) were 57%, 50%, 87% and 29%, respectively. FVC increased at a rate of 1.8% per year (P = 0.008). There was no significant change in FEV1 over time (P = 0.112). However, the FEV1/FVC ratio decreased by 2.6% per year (P &lt; 0.001). Conclusion PFT in childhood PBO was characterized by significant airway obstruction. Over time, FVC (lung parenchyma) increased and FEV1 (airway) remained stable, but FEV1/FVC ratio declined more than expected, suggesting a mismatch in the growth of the airway and lung parenchyma (dysanaptic growth). Further studies in larger populations are needed to validate these observations.</description><subject>Adolescent</subject><subject>Airway management</subject><subject>bronchiolitis obliterans</subject><subject>Bronchiolitis Obliterans - diagnostic imaging</subject><subject>Bronchiolitis Obliterans - etiology</subject><subject>Bronchiolitis Obliterans - pathology</subject><subject>Bronchiolitis Obliterans - physiopathology</subject><subject>Child</subject><subject>Disease Progression</subject><subject>dysanaptic growth</subject><subject>Female</subject><subject>Forced Expiratory Volume</subject><subject>Humans</subject><subject>immunomodulation</subject><subject>Inflammation - pathology</subject><subject>Inflammation - physiopathology</subject><subject>inhaled corticosteroids</subject><subject>Lung - pathology</subject><subject>Lung - physiopathology</subject><subject>Male</subject><subject>pulmonary function test</subject><subject>Respiratory diseases</subject><subject>Respiratory Function Tests</subject><subject>Respiratory Tract Infections - complications</subject><subject>Tomography, X-Ray Computed - methods</subject><subject>Vital Capacity</subject><issn>1752-6981</issn><issn>1752-699X</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2014</creationdate><recordtype>article</recordtype><recordid>eNp1kE2P0zAQhi3EinYLB_4AssQFDunacRwnR1SWAqp20VIE4oDlOE7rktpZO1G3_55h-3FAwj6MD8-8M34QeknJlMK50mEzpSlh4gkaU8HTJC_LH0_P74KO0GWMG0J4IRh_hkYpKwhJSTFGv97vo3Kq663Gq-B3_Rr7BreDW2Hlaqxs2Kk9tg7rtW3rYBzeWWA6H_vEusbo3voh4ip4B4RvbW8j9hVUE5SLz9FFo9poXhzrBH37cL2cfUwWt_NPs3eLRLOSiiRLVcV0Xhe5EKpQdWMoMTnj1DCT0zKtSQVcQeBS0bCqLGoNs_NaVDTjXLEJenPI7YK_H0zs5dZGbdpWOQP7SZqVRDABXgB9_Q-68UNwsB1QIst4yUDlBL09UDr4GINpZBfsVoW9pET-lS5BunyUDuyrY-JQbU19Jk-WAbg6ADvbmv3_k-Ts7vMpMjl02Nibh3OHCr9lDv_g8vvNXM6-LDn5Se7kV_YHg02a7w</recordid><startdate>201401</startdate><enddate>201401</enddate><creator>Mosquera, Ricardo A.</creator><creator>Hashmi, Syed S.</creator><creator>Pacheco, Susan E.</creator><creator>Reverdin, Alexandra</creator><creator>Chevallier, Justyna</creator><creator>Colasurdo, Giuseppe N.</creator><general>Blackwell Publishing Ltd</general><general>John Wiley &amp; Sons, Inc</general><scope>BSCLL</scope><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>7X8</scope></search><sort><creationdate>201401</creationdate><title>Dysanaptic growth of lung and airway in children with post-infectious bronchiolitis obliterans</title><author>Mosquera, Ricardo A. ; Hashmi, Syed S. ; Pacheco, Susan E. ; Reverdin, Alexandra ; Chevallier, Justyna ; Colasurdo, Giuseppe N.</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c3917-42ab3c6d8677a8adfe10e6351e3e6192d0b3918080817f3b98dcfec6d7b1455a3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2014</creationdate><topic>Adolescent</topic><topic>Airway management</topic><topic>bronchiolitis obliterans</topic><topic>Bronchiolitis Obliterans - diagnostic imaging</topic><topic>Bronchiolitis Obliterans - etiology</topic><topic>Bronchiolitis Obliterans - pathology</topic><topic>Bronchiolitis Obliterans - physiopathology</topic><topic>Child</topic><topic>Disease Progression</topic><topic>dysanaptic growth</topic><topic>Female</topic><topic>Forced Expiratory Volume</topic><topic>Humans</topic><topic>immunomodulation</topic><topic>Inflammation - pathology</topic><topic>Inflammation - physiopathology</topic><topic>inhaled corticosteroids</topic><topic>Lung - pathology</topic><topic>Lung - physiopathology</topic><topic>Male</topic><topic>pulmonary function test</topic><topic>Respiratory diseases</topic><topic>Respiratory Function Tests</topic><topic>Respiratory Tract Infections - complications</topic><topic>Tomography, X-Ray Computed - methods</topic><topic>Vital Capacity</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Mosquera, Ricardo A.</creatorcontrib><creatorcontrib>Hashmi, Syed S.</creatorcontrib><creatorcontrib>Pacheco, Susan E.</creatorcontrib><creatorcontrib>Reverdin, Alexandra</creatorcontrib><creatorcontrib>Chevallier, Justyna</creatorcontrib><creatorcontrib>Colasurdo, Giuseppe N.</creatorcontrib><collection>Istex</collection><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 &amp; Medical Complete (Alumni)</collection><collection>MEDLINE - Academic</collection><jtitle>The clinical respiratory journal</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Mosquera, Ricardo A.</au><au>Hashmi, Syed S.</au><au>Pacheco, Susan E.</au><au>Reverdin, Alexandra</au><au>Chevallier, Justyna</au><au>Colasurdo, Giuseppe N.</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Dysanaptic growth of lung and airway in children with post-infectious bronchiolitis obliterans</atitle><jtitle>The clinical respiratory journal</jtitle><addtitle>The Clinical Respiratory Journal</addtitle><date>2014-01</date><risdate>2014</risdate><volume>8</volume><issue>1</issue><spage>63</spage><epage>71</epage><pages>63-71</pages><issn>1752-6981</issn><eissn>1752-699X</eissn><notes>ark:/67375/WNG-CPT50Z0R-S</notes><notes>istex:94D351A0A2E8EB0B22FE18EEEEE90FF48E0A4664</notes><notes>ArticleID:CRJ12037</notes><notes>Ethics</notes><notes>The authors have disclosed no potential conflicts of interest. No funding was obtained.</notes><notes>Authorship and contributorship</notes><notes>All authors listed meet definition of authorship.</notes><notes>Conflict of interest</notes><notes>The study was reviewed and approved by the University of Texas Medical School at Houston institutional review board prior to initiation of this retrospective chart review.</notes><notes>ObjectType-Article-1</notes><notes>SourceType-Scholarly Journals-1</notes><notes>ObjectType-Feature-2</notes><notes>content type line 23</notes><abstract>Rationale Post‐infectious bronchiolitis obliterans (PBO) is a rare form of chronic obstructive lung disease associated with small airway fibrosis following a severe insult to the lower respiratory tract. It has been suggested that PBO is a non‐progressive disease. However, evidence supporting this statement is limited. In this case series, we sought to determine the changes of pulmonary function tests (PFT) over time in children with PBO. Methods Seven children with PBO, ages 6–15 years old, were retrospectively studied between 1994 and 2012. Spirometry and lung volumes tests were performed in accordance with American Thoracic Society (ATS) guidelines and were monitored over time. The average rate of change was calculated using generalized linear mixed models. Results The median baseline values for forced vital capacity (FVC), forced expiratory volume in 1 s (FEV1), the FEV1/FVC ratio and forced expiratory flow 25%–75% (FEF25%–75%) were 57%, 50%, 87% and 29%, respectively. FVC increased at a rate of 1.8% per year (P = 0.008). There was no significant change in FEV1 over time (P = 0.112). However, the FEV1/FVC ratio decreased by 2.6% per year (P &lt; 0.001). Conclusion PFT in childhood PBO was characterized by significant airway obstruction. Over time, FVC (lung parenchyma) increased and FEV1 (airway) remained stable, but FEV1/FVC ratio declined more than expected, suggesting a mismatch in the growth of the airway and lung parenchyma (dysanaptic growth). Further studies in larger populations are needed to validate these observations.</abstract><cop>England</cop><pub>Blackwell Publishing Ltd</pub><pmid>23800208</pmid><doi>10.1111/crj.12037</doi><tpages>9</tpages></addata></record>
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subjects Adolescent
Airway management
bronchiolitis obliterans
Bronchiolitis Obliterans - diagnostic imaging
Bronchiolitis Obliterans - etiology
Bronchiolitis Obliterans - pathology
Bronchiolitis Obliterans - physiopathology
Child
Disease Progression
dysanaptic growth
Female
Forced Expiratory Volume
Humans
immunomodulation
Inflammation - pathology
Inflammation - physiopathology
inhaled corticosteroids
Lung - pathology
Lung - physiopathology
Male
pulmonary function test
Respiratory diseases
Respiratory Function Tests
Respiratory Tract Infections - complications
Tomography, X-Ray Computed - methods
Vital Capacity
title Dysanaptic growth of lung and airway in children with post-infectious bronchiolitis obliterans
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