Serum KL-6 differentiates neuroendocrine cell hyperplasia of infancy from the inborn errors of surfactant metabolism
Background:The study was conducted in order to determine if the glycoprotein KL-6 is a useful biomarker in differentiating neuroendocrine cell hyperplasia of infancy (NEHI), a benign form of children’s interstitial lung disease, from the more severe inborn errors of surfactant metabolism (IESM), sin...
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| Veröffentlicht in: | Thorax 2009-08, Vol.64 (8), p.677-681 |
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| creator | Doan, M L Elidemir, O Dishop, M K Zhang, H Smith, E O Black, P G Deterding, R R Roberts, D M Al-Salmi, Q A Fan, L L |
| description | Background:The study was conducted in order to determine if the glycoprotein KL-6 is a useful biomarker in differentiating neuroendocrine cell hyperplasia of infancy (NEHI), a benign form of children’s interstitial lung disease, from the more severe inborn errors of surfactant metabolism (IESM), since their clinical presentation can be similar.Methods:Serum KL-6 levels were measured in 10 healthy control children, 6 with NEHI and 13 with IESM (4 with surfactant protein C (SP-C) and 9 with ABCA3 mutations). The initial clinical presentation, findings on previous CT scans and interstitial lung disease (ILD) scores at the time of KL-6 testing were compared. Correlations of KL-6 levels with age and with interval from lung biopsy were evaluated.Results:The median (range) KL-6 levels were 265 (1–409), 194 (47–352), 1149 (593–4407) and 3068 (726–9912) U/ml for the control, NEHI, SP-C and ABCA3 groups, respectively. When compared with the control and NEHI groups, median KL-6 levels were significantly higher in the SP-C (p |
| doi_str_mv | 10.1136/thx.2008.107979 |
| format | Article |
| fullrecord | <record><control><sourceid>proquest_cross</sourceid><recordid>TN_cdi_proquest_miscellaneous_733573741</recordid><sourceformat>XML</sourceformat><sourcesystem>PC</sourcesystem><sourcerecordid>4026982191</sourcerecordid><originalsourceid>FETCH-LOGICAL-b468t-e8fb19938438e77211194c0dfbd325502cd91e451de5b8c84add6d49ea465c8e3</originalsourceid><addsrcrecordid>eNqF0d-rFCEUB3CJorttPfcWQkQQzF4dZ9R5jKVftNVDP-6jOM6RdZvRverA3f8-l1lu0EsgCPrxcI5fhJ5TsqGU8eu8v9vUhMgNJaIT3QO0og2XFas7_hCtCGlIxZngV-hJSgdSIKXiMbqiXc0Ek90K5e8Q5wl_3lUcD85aiOCz0xkS9jDHAH4IJjoP2MA44v3pCPE46uQ0DhY7b7U3J2xjmHDeQznoQ_QYYgwxnUWao9Uma5_xBFn3YXRpeooeWT0meHbZ1-jn-3c_th-r3bcPn7Zvd1VfhsgVSNvTrmOyYRKEqCmlXWPIYPuB1W1LajN0FJqWDtD20shGDwMfmg50w1sjga3R66XuMYbbGVJWk0vnObSHMCclGGsFEw0t8uU_8hDm6EtzigpZFqecFHW9KBNDShGsOkY36XhSlKhzHqrkoc55qCWP8uLFpe7cTzD89ZcACnh1AToZPdpY_tOle1dTUcvSY3HV4lzKcHd_r-NvxcsIrfr6a6uadnvzhdzUZbA1erP4fjr8t8s_ym-wfQ</addsrcrecordid><sourcetype>Aggregation Database</sourcetype><iscdi>true</iscdi><recordtype>article</recordtype><pqid>1781786160</pqid></control><display><type>article</type><title>Serum KL-6 differentiates neuroendocrine cell hyperplasia of infancy from the inborn errors of surfactant metabolism</title><source>MEDLINE</source><source>BMJ Journals - NESLi2</source><source>Alma/SFX Local Collection</source><creator>Doan, M L ; Elidemir, O ; Dishop, M K ; Zhang, H ; Smith, E O ; Black, P G ; Deterding, R R ; Roberts, D M ; Al-Salmi, Q A ; Fan, L L</creator><creatorcontrib>Doan, M L ; Elidemir, O ; Dishop, M K ; Zhang, H ; Smith, E O ; Black, P G ; Deterding, R R ; Roberts, D M ; Al-Salmi, Q A ; Fan, L L</creatorcontrib><description>Background:The study was conducted in order to determine if the glycoprotein KL-6 is a useful biomarker in differentiating neuroendocrine cell hyperplasia of infancy (NEHI), a benign form of children’s interstitial lung disease, from the more severe inborn errors of surfactant metabolism (IESM), since their clinical presentation can be similar.Methods:Serum KL-6 levels were measured in 10 healthy control children, 6 with NEHI and 13 with IESM (4 with surfactant protein C (SP-C) and 9 with ABCA3 mutations). The initial clinical presentation, findings on previous CT scans and interstitial lung disease (ILD) scores at the time of KL-6 testing were compared. Correlations of KL-6 levels with age and with interval from lung biopsy were evaluated.Results:The median (range) KL-6 levels were 265 (1–409), 194 (47–352), 1149 (593–4407) and 3068 (726–9912) U/ml for the control, NEHI, SP-C and ABCA3 groups, respectively. When compared with the control and NEHI groups, median KL-6 levels were significantly higher in the SP-C (p<0.01; p = 0.01, respectively) and ABCA3 groups (p<0.001; p = 0.001, respectively); however, there was no difference between the control and NEHI groups (p = 0.91). An inverse relationship was seen between KL-6 levels and age in the IESM groups, but not in the NEHI or control groups. Children with NEHI had similar presenting clinical features and were equally symptomatic at the time of KL-6 measurement as those with IESM.Conclusions:Children with NEHI have normal KL-6 levels, in contrast to those with IESM, who have elevated serum KL-6 levels; serum KL-6 may be a useful biomarker in distinguishing between these entities when their clinical presentations overlap.</description><identifier>ISSN: 0040-6376</identifier><identifier>EISSN: 1468-3296</identifier><identifier>DOI: 10.1136/thx.2008.107979</identifier><identifier>PMID: 19237389</identifier><identifier>CODEN: THORA7</identifier><language>eng</language><publisher>London: BMJ Publishing Group Ltd and British Thoracic Society</publisher><subject>Adolescent ; ATP-Binding Cassette Transporters - genetics ; Biological and medical sciences ; Biomarkers ; Biomarkers - metabolism ; Biopsy ; Cardiology. Vascular system ; Child ; Child, Preschool ; Children & youth ; Histology ; Humans ; Hyperplasia ; Hyperplasia - metabolism ; Hyperplasia - pathology ; Infant ; Inflammation ; Laboratories ; Lipid Metabolism, Inborn Errors ; Lung - metabolism ; Lung - pathology ; Lung diseases ; Lung Diseases, Interstitial - metabolism ; Lung Diseases, Interstitial - pathology ; Medical sciences ; Metabolism ; Mucin-1 - metabolism ; Mutation ; Neuroendocrine Cells - metabolism ; Neuroendocrine Cells - pathology ; Pneumology ; Pneumonia ; Pulmonary Surfactant-Associated Protein C - genetics ; Pulmonary Surfactant-Associated Protein C - metabolism ; Respiratory failure ; Steroids ; Surfactants</subject><ispartof>Thorax, 2009-08, Vol.64 (8), p.677-681</ispartof><rights>2009 BMJ Publishing Group Ltd and the British Thoracic Society</rights><rights>2009 INIST-CNRS</rights><rights>Copyright: 2009 2009 BMJ Publishing Group Ltd and the British Thoracic Society</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-b468t-e8fb19938438e77211194c0dfbd325502cd91e451de5b8c84add6d49ea465c8e3</citedby></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://thorax.bmj.com/content/64/8/677.full.pdf$$EPDF$$P50$$Gbmj$$H</linktopdf><linktohtml>$$Uhttps://thorax.bmj.com/content/64/8/677.full$$EHTML$$P50$$Gbmj$$H</linktohtml><link.rule.ids>114,115,315,782,786,3198,23578,27931,27932,77608,77639</link.rule.ids><backlink>$$Uhttp://pascal-francis.inist.fr/vibad/index.php?action=getRecordDetail&idt=21728573$$DView record in Pascal Francis$$Hfree_for_read</backlink><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/19237389$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Doan, M L</creatorcontrib><creatorcontrib>Elidemir, O</creatorcontrib><creatorcontrib>Dishop, M K</creatorcontrib><creatorcontrib>Zhang, H</creatorcontrib><creatorcontrib>Smith, E O</creatorcontrib><creatorcontrib>Black, P G</creatorcontrib><creatorcontrib>Deterding, R R</creatorcontrib><creatorcontrib>Roberts, D M</creatorcontrib><creatorcontrib>Al-Salmi, Q A</creatorcontrib><creatorcontrib>Fan, L L</creatorcontrib><title>Serum KL-6 differentiates neuroendocrine cell hyperplasia of infancy from the inborn errors of surfactant metabolism</title><title>Thorax</title><addtitle>Thorax</addtitle><description>Background:The study was conducted in order to determine if the glycoprotein KL-6 is a useful biomarker in differentiating neuroendocrine cell hyperplasia of infancy (NEHI), a benign form of children’s interstitial lung disease, from the more severe inborn errors of surfactant metabolism (IESM), since their clinical presentation can be similar.Methods:Serum KL-6 levels were measured in 10 healthy control children, 6 with NEHI and 13 with IESM (4 with surfactant protein C (SP-C) and 9 with ABCA3 mutations). The initial clinical presentation, findings on previous CT scans and interstitial lung disease (ILD) scores at the time of KL-6 testing were compared. Correlations of KL-6 levels with age and with interval from lung biopsy were evaluated.Results:The median (range) KL-6 levels were 265 (1–409), 194 (47–352), 1149 (593–4407) and 3068 (726–9912) U/ml for the control, NEHI, SP-C and ABCA3 groups, respectively. When compared with the control and NEHI groups, median KL-6 levels were significantly higher in the SP-C (p<0.01; p = 0.01, respectively) and ABCA3 groups (p<0.001; p = 0.001, respectively); however, there was no difference between the control and NEHI groups (p = 0.91). An inverse relationship was seen between KL-6 levels and age in the IESM groups, but not in the NEHI or control groups. Children with NEHI had similar presenting clinical features and were equally symptomatic at the time of KL-6 measurement as those with IESM.Conclusions:Children with NEHI have normal KL-6 levels, in contrast to those with IESM, who have elevated serum KL-6 levels; serum KL-6 may be a useful biomarker in distinguishing between these entities when their clinical presentations overlap.</description><subject>Adolescent</subject><subject>ATP-Binding Cassette Transporters - genetics</subject><subject>Biological and medical sciences</subject><subject>Biomarkers</subject><subject>Biomarkers - metabolism</subject><subject>Biopsy</subject><subject>Cardiology. Vascular system</subject><subject>Child</subject><subject>Child, Preschool</subject><subject>Children & youth</subject><subject>Histology</subject><subject>Humans</subject><subject>Hyperplasia</subject><subject>Hyperplasia - metabolism</subject><subject>Hyperplasia - pathology</subject><subject>Infant</subject><subject>Inflammation</subject><subject>Laboratories</subject><subject>Lipid Metabolism, Inborn Errors</subject><subject>Lung - metabolism</subject><subject>Lung - pathology</subject><subject>Lung diseases</subject><subject>Lung Diseases, Interstitial - metabolism</subject><subject>Lung Diseases, Interstitial - pathology</subject><subject>Medical sciences</subject><subject>Metabolism</subject><subject>Mucin-1 - metabolism</subject><subject>Mutation</subject><subject>Neuroendocrine Cells - metabolism</subject><subject>Neuroendocrine Cells - pathology</subject><subject>Pneumology</subject><subject>Pneumonia</subject><subject>Pulmonary Surfactant-Associated Protein C - genetics</subject><subject>Pulmonary Surfactant-Associated Protein C - metabolism</subject><subject>Respiratory failure</subject><subject>Steroids</subject><subject>Surfactants</subject><issn>0040-6376</issn><issn>1468-3296</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2009</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><sourceid>ABUWG</sourceid><sourceid>AFKRA</sourceid><sourceid>BENPR</sourceid><sourceid>CCPQU</sourceid><recordid>eNqF0d-rFCEUB3CJorttPfcWQkQQzF4dZ9R5jKVftNVDP-6jOM6RdZvRverA3f8-l1lu0EsgCPrxcI5fhJ5TsqGU8eu8v9vUhMgNJaIT3QO0og2XFas7_hCtCGlIxZngV-hJSgdSIKXiMbqiXc0Ek90K5e8Q5wl_3lUcD85aiOCz0xkS9jDHAH4IJjoP2MA44v3pCPE46uQ0DhY7b7U3J2xjmHDeQznoQ_QYYgwxnUWao9Uma5_xBFn3YXRpeooeWT0meHbZ1-jn-3c_th-r3bcPn7Zvd1VfhsgVSNvTrmOyYRKEqCmlXWPIYPuB1W1LajN0FJqWDtD20shGDwMfmg50w1sjga3R66XuMYbbGVJWk0vnObSHMCclGGsFEw0t8uU_8hDm6EtzigpZFqecFHW9KBNDShGsOkY36XhSlKhzHqrkoc55qCWP8uLFpe7cTzD89ZcACnh1AToZPdpY_tOle1dTUcvSY3HV4lzKcHd_r-NvxcsIrfr6a6uadnvzhdzUZbA1erP4fjr8t8s_ym-wfQ</recordid><startdate>20090801</startdate><enddate>20090801</enddate><creator>Doan, M L</creator><creator>Elidemir, O</creator><creator>Dishop, M K</creator><creator>Zhang, H</creator><creator>Smith, E O</creator><creator>Black, P G</creator><creator>Deterding, R R</creator><creator>Roberts, D M</creator><creator>Al-Salmi, Q A</creator><creator>Fan, L L</creator><general>BMJ Publishing Group Ltd and British Thoracic Society</general><general>BMJ Publishing Group</general><general>BMJ Publishing Group LTD</general><scope>BSCLL</scope><scope>IQODW</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>3V.</scope><scope>7X7</scope><scope>7XB</scope><scope>88E</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>ABUWG</scope><scope>AFKRA</scope><scope>BENPR</scope><scope>BTHHO</scope><scope>CCPQU</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>K9.</scope><scope>M0S</scope><scope>M1P</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>PRINS</scope><scope>7X8</scope></search><sort><creationdate>20090801</creationdate><title>Serum KL-6 differentiates neuroendocrine cell hyperplasia of infancy from the inborn errors of surfactant metabolism</title><author>Doan, M L ; Elidemir, O ; Dishop, M K ; Zhang, H ; Smith, E O ; Black, P G ; Deterding, R R ; Roberts, D M ; Al-Salmi, Q A ; Fan, L L</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-b468t-e8fb19938438e77211194c0dfbd325502cd91e451de5b8c84add6d49ea465c8e3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2009</creationdate><topic>Adolescent</topic><topic>ATP-Binding Cassette Transporters - genetics</topic><topic>Biological and medical sciences</topic><topic>Biomarkers</topic><topic>Biomarkers - metabolism</topic><topic>Biopsy</topic><topic>Cardiology. Vascular system</topic><topic>Child</topic><topic>Child, Preschool</topic><topic>Children & youth</topic><topic>Histology</topic><topic>Humans</topic><topic>Hyperplasia</topic><topic>Hyperplasia - metabolism</topic><topic>Hyperplasia - pathology</topic><topic>Infant</topic><topic>Inflammation</topic><topic>Laboratories</topic><topic>Lipid Metabolism, Inborn Errors</topic><topic>Lung - metabolism</topic><topic>Lung - pathology</topic><topic>Lung diseases</topic><topic>Lung Diseases, Interstitial - metabolism</topic><topic>Lung Diseases, Interstitial - pathology</topic><topic>Medical sciences</topic><topic>Metabolism</topic><topic>Mucin-1 - metabolism</topic><topic>Mutation</topic><topic>Neuroendocrine Cells - metabolism</topic><topic>Neuroendocrine Cells - pathology</topic><topic>Pneumology</topic><topic>Pneumonia</topic><topic>Pulmonary Surfactant-Associated Protein C - genetics</topic><topic>Pulmonary Surfactant-Associated Protein C - metabolism</topic><topic>Respiratory failure</topic><topic>Steroids</topic><topic>Surfactants</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Doan, M L</creatorcontrib><creatorcontrib>Elidemir, O</creatorcontrib><creatorcontrib>Dishop, M K</creatorcontrib><creatorcontrib>Zhang, H</creatorcontrib><creatorcontrib>Smith, E O</creatorcontrib><creatorcontrib>Black, P G</creatorcontrib><creatorcontrib>Deterding, R R</creatorcontrib><creatorcontrib>Roberts, D M</creatorcontrib><creatorcontrib>Al-Salmi, Q A</creatorcontrib><creatorcontrib>Fan, L L</creatorcontrib><collection>Istex</collection><collection>Pascal-Francis</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 Central (Corporate)</collection><collection>Health & Medical Collection</collection><collection>ProQuest Central (purchase pre-March 2016)</collection><collection>Medical Database (Alumni Edition)</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 Edition)</collection><collection>ProQuest Central UK/Ireland</collection><collection>ProQuest Central</collection><collection>BMJ Journals</collection><collection>ProQuest One Community College</collection><collection>Health Research Premium Collection</collection><collection>Health Research Premium Collection (Alumni)</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>Health & Medical Collection (Alumni Edition)</collection><collection>Medical Database</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 China</collection><collection>MEDLINE - Academic</collection><jtitle>Thorax</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Doan, M L</au><au>Elidemir, O</au><au>Dishop, M K</au><au>Zhang, H</au><au>Smith, E O</au><au>Black, P G</au><au>Deterding, R R</au><au>Roberts, D M</au><au>Al-Salmi, Q A</au><au>Fan, L L</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Serum KL-6 differentiates neuroendocrine cell hyperplasia of infancy from the inborn errors of surfactant metabolism</atitle><jtitle>Thorax</jtitle><addtitle>Thorax</addtitle><date>2009-08-01</date><risdate>2009</risdate><volume>64</volume><issue>8</issue><spage>677</spage><epage>681</epage><pages>677-681</pages><issn>0040-6376</issn><eissn>1468-3296</eissn><coden>THORA7</coden><abstract>Background:The study was conducted in order to determine if the glycoprotein KL-6 is a useful biomarker in differentiating neuroendocrine cell hyperplasia of infancy (NEHI), a benign form of children’s interstitial lung disease, from the more severe inborn errors of surfactant metabolism (IESM), since their clinical presentation can be similar.Methods:Serum KL-6 levels were measured in 10 healthy control children, 6 with NEHI and 13 with IESM (4 with surfactant protein C (SP-C) and 9 with ABCA3 mutations). The initial clinical presentation, findings on previous CT scans and interstitial lung disease (ILD) scores at the time of KL-6 testing were compared. Correlations of KL-6 levels with age and with interval from lung biopsy were evaluated.Results:The median (range) KL-6 levels were 265 (1–409), 194 (47–352), 1149 (593–4407) and 3068 (726–9912) U/ml for the control, NEHI, SP-C and ABCA3 groups, respectively. When compared with the control and NEHI groups, median KL-6 levels were significantly higher in the SP-C (p<0.01; p = 0.01, respectively) and ABCA3 groups (p<0.001; p = 0.001, respectively); however, there was no difference between the control and NEHI groups (p = 0.91). An inverse relationship was seen between KL-6 levels and age in the IESM groups, but not in the NEHI or control groups. Children with NEHI had similar presenting clinical features and were equally symptomatic at the time of KL-6 measurement as those with IESM.Conclusions:Children with NEHI have normal KL-6 levels, in contrast to those with IESM, who have elevated serum KL-6 levels; serum KL-6 may be a useful biomarker in distinguishing between these entities when their clinical presentations overlap.</abstract><cop>London</cop><pub>BMJ Publishing Group Ltd and British Thoracic Society</pub><pmid>19237389</pmid><doi>10.1136/thx.2008.107979</doi><tpages>5</tpages><oa>free_for_read</oa></addata></record> |
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| subjects | Adolescent ATP-Binding Cassette Transporters - genetics Biological and medical sciences Biomarkers Biomarkers - metabolism Biopsy Cardiology. Vascular system Child Child, Preschool Children & youth Histology Humans Hyperplasia Hyperplasia - metabolism Hyperplasia - pathology Infant Inflammation Laboratories Lipid Metabolism, Inborn Errors Lung - metabolism Lung - pathology Lung diseases Lung Diseases, Interstitial - metabolism Lung Diseases, Interstitial - pathology Medical sciences Metabolism Mucin-1 - metabolism Mutation Neuroendocrine Cells - metabolism Neuroendocrine Cells - pathology Pneumology Pneumonia Pulmonary Surfactant-Associated Protein C - genetics Pulmonary Surfactant-Associated Protein C - metabolism Respiratory failure Steroids Surfactants |
| title | Serum KL-6 differentiates neuroendocrine cell hyperplasia of infancy from the inborn errors of surfactant metabolism |
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