Cytomegalovirus-associated biliary atresia: An aetiological and prognostic subgroup

Abstract Background and aims Perinatal cytomegalovirus (CMV) infection is a possible cause or trigger of biliary atresia though clinical evidence is scant. We hypothesised that CMV IgM+ve biliary atresia is a separate clinical entity compared to CMV IgM−ve biliary atresia. Methods Prospective single...

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Veröffentlicht in:Journal of pediatric surgery 2015-10, Vol.50 (10), p.1739-1745
Hauptverfasser: Zani, Augusto, Quaglia, Alberto, Hadzić, Nedim, Zuckerman, Mark, Davenport, Mark
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container_end_page 1745
container_issue 10
container_start_page 1739
container_title Journal of pediatric surgery
container_volume 50
creator Zani, Augusto
Quaglia, Alberto
Hadzić, Nedim
Zuckerman, Mark
Davenport, Mark
description Abstract Background and aims Perinatal cytomegalovirus (CMV) infection is a possible cause or trigger of biliary atresia though clinical evidence is scant. We hypothesised that CMV IgM+ve biliary atresia is a separate clinical entity compared to CMV IgM−ve biliary atresia. Methods Prospective single-centre study. 210 infants with histologically confirmed biliary atresia were treated in our institution (Jan. 2004 to Dec. 2011); of these 20 (9.5%) were CMV IgM+ve at presentation. We compared these with 111 infants who were CMV IgM−ve (controls) for clinical features, biochemistry at presentation and outcome following Kasai portoenterostomy (KPE). A blinded comparison of age-matched liver histology was also performed. Data are quoted as median (interquartile range). A P value ≤ 0.05 was regarded as significant. Results Infants with CMV IgM+ve biliary atresia were older at Kasai portoenterostomy (or laparotomy) [70 (60–80) days vs. 56 (44–75)days; P = 0.003] and were more jaundiced [175 (147–224) vs. 140 (121–181) μmol/L; P = 0.002 + with higher AST*287 (157–403) vs. 180 (133–254) IU/L; P = 0.005] and aspartate aminotransferase-to-platelet ratio index [1.1 (0.79–3.0) vs. 0.63 (0.43–0.95)] levels. Liver histology : CMV IgM+ve biliary atresia was characterised by a greater degree of inflammation (P < 0.0001) and fibrosis (P = 0.02), whereas CMV IgM−ve isolated biliary atresia had a higher degree of lobular cholestasis (P = 0.001). This effect was independent of the effects of age at KPE. Outcome : CMV IgM+ve biliary atresia had a poorer outcome with a reduced clearance of jaundice (15% vs. 52.2%; P = 0.002), native liver survival (P < 0.0001) and increased mortality (P = 0.002). Conclusions CMV IgM+ve biliary atresia is a distinct clinical and pathological entity with a diminished response to Kasai portoenterostomy.
doi_str_mv 10.1016/j.jpedsurg.2015.03.001
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We hypothesised that CMV IgM+ve biliary atresia is a separate clinical entity compared to CMV IgM−ve biliary atresia. Methods Prospective single-centre study. 210 infants with histologically confirmed biliary atresia were treated in our institution (Jan. 2004 to Dec. 2011); of these 20 (9.5%) were CMV IgM+ve at presentation. We compared these with 111 infants who were CMV IgM−ve (controls) for clinical features, biochemistry at presentation and outcome following Kasai portoenterostomy (KPE). A blinded comparison of age-matched liver histology was also performed. Data are quoted as median (interquartile range). A P value ≤ 0.05 was regarded as significant. Results Infants with CMV IgM+ve biliary atresia were older at Kasai portoenterostomy (or laparotomy) [70 (60–80) days vs. 56 (44–75)days; P = 0.003] and were more jaundiced [175 (147–224) vs. 140 (121–181) μmol/L; P = 0.002 + with higher AST*287 (157–403) vs. 180 (133–254) IU/L; P = 0.005] and aspartate aminotransferase-to-platelet ratio index [1.1 (0.79–3.0) vs. 0.63 (0.43–0.95)] levels. Liver histology : CMV IgM+ve biliary atresia was characterised by a greater degree of inflammation (P &lt; 0.0001) and fibrosis (P = 0.02), whereas CMV IgM−ve isolated biliary atresia had a higher degree of lobular cholestasis (P = 0.001). This effect was independent of the effects of age at KPE. Outcome : CMV IgM+ve biliary atresia had a poorer outcome with a reduced clearance of jaundice (15% vs. 52.2%; P = 0.002), native liver survival (P &lt; 0.0001) and increased mortality (P = 0.002). Conclusions CMV IgM+ve biliary atresia is a distinct clinical and pathological entity with a diminished response to Kasai portoenterostomy.</description><identifier>ISSN: 0022-3468</identifier><identifier>EISSN: 1531-5037</identifier><identifier>DOI: 10.1016/j.jpedsurg.2015.03.001</identifier><identifier>PMID: 25824438</identifier><language>eng</language><publisher>United States: Elsevier Inc</publisher><subject>Age Factors ; Aspartate Aminotransferases - blood ; Biliary atresia ; Biliary Atresia - pathology ; Biliary Atresia - surgery ; Biliary Atresia - virology ; Cholestasis - etiology ; Cytomegalovirus (CMV) ; Cytomegalovirus - immunology ; Cytomegalovirus Infections - complications ; Female ; Humans ; Immunoglobulin M - analysis ; Infant ; Kasai portoenterostomy ; Liver - enzymology ; Liver - pathology ; Liver - surgery ; Male ; Neonatal cholestatic jaundice ; Pediatrics ; Platelet Count ; Portoenterostomy, Hepatic ; Prognosis ; Prospective Studies ; Surgery</subject><ispartof>Journal of pediatric surgery, 2015-10, Vol.50 (10), p.1739-1745</ispartof><rights>Elsevier Inc.</rights><rights>2015 Elsevier Inc.</rights><rights>Copyright © 2015 Elsevier Inc. All rights reserved.</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c629t-c7017847aa853df9b21a79221eb81f82911c5a5c219c61b3e8a5c095c69926803</citedby><cites>FETCH-LOGICAL-c629t-c7017847aa853df9b21a79221eb81f82911c5a5c219c61b3e8a5c095c69926803</cites><orcidid>0000-0002-5493-3418</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktohtml>$$Uhttps://www.sciencedirect.com/science/article/pii/S0022346815001633$$EHTML$$P50$$Gelsevier$$H</linktohtml><link.rule.ids>314,776,780,3537,27901,27902,65306</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/25824438$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Zani, Augusto</creatorcontrib><creatorcontrib>Quaglia, Alberto</creatorcontrib><creatorcontrib>Hadzić, Nedim</creatorcontrib><creatorcontrib>Zuckerman, Mark</creatorcontrib><creatorcontrib>Davenport, Mark</creatorcontrib><title>Cytomegalovirus-associated biliary atresia: An aetiological and prognostic subgroup</title><title>Journal of pediatric surgery</title><addtitle>J Pediatr Surg</addtitle><description>Abstract Background and aims Perinatal cytomegalovirus (CMV) infection is a possible cause or trigger of biliary atresia though clinical evidence is scant. We hypothesised that CMV IgM+ve biliary atresia is a separate clinical entity compared to CMV IgM−ve biliary atresia. Methods Prospective single-centre study. 210 infants with histologically confirmed biliary atresia were treated in our institution (Jan. 2004 to Dec. 2011); of these 20 (9.5%) were CMV IgM+ve at presentation. We compared these with 111 infants who were CMV IgM−ve (controls) for clinical features, biochemistry at presentation and outcome following Kasai portoenterostomy (KPE). A blinded comparison of age-matched liver histology was also performed. Data are quoted as median (interquartile range). A P value ≤ 0.05 was regarded as significant. Results Infants with CMV IgM+ve biliary atresia were older at Kasai portoenterostomy (or laparotomy) [70 (60–80) days vs. 56 (44–75)days; P = 0.003] and were more jaundiced [175 (147–224) vs. 140 (121–181) μmol/L; P = 0.002 + with higher AST*287 (157–403) vs. 180 (133–254) IU/L; P = 0.005] and aspartate aminotransferase-to-platelet ratio index [1.1 (0.79–3.0) vs. 0.63 (0.43–0.95)] levels. Liver histology : CMV IgM+ve biliary atresia was characterised by a greater degree of inflammation (P &lt; 0.0001) and fibrosis (P = 0.02), whereas CMV IgM−ve isolated biliary atresia had a higher degree of lobular cholestasis (P = 0.001). This effect was independent of the effects of age at KPE. Outcome : CMV IgM+ve biliary atresia had a poorer outcome with a reduced clearance of jaundice (15% vs. 52.2%; P = 0.002), native liver survival (P &lt; 0.0001) and increased mortality (P = 0.002). Conclusions CMV IgM+ve biliary atresia is a distinct clinical and pathological entity with a diminished response to Kasai portoenterostomy.</description><subject>Age Factors</subject><subject>Aspartate Aminotransferases - blood</subject><subject>Biliary atresia</subject><subject>Biliary Atresia - pathology</subject><subject>Biliary Atresia - surgery</subject><subject>Biliary Atresia - virology</subject><subject>Cholestasis - etiology</subject><subject>Cytomegalovirus (CMV)</subject><subject>Cytomegalovirus - immunology</subject><subject>Cytomegalovirus Infections - complications</subject><subject>Female</subject><subject>Humans</subject><subject>Immunoglobulin M - analysis</subject><subject>Infant</subject><subject>Kasai portoenterostomy</subject><subject>Liver - enzymology</subject><subject>Liver - pathology</subject><subject>Liver - surgery</subject><subject>Male</subject><subject>Neonatal cholestatic jaundice</subject><subject>Pediatrics</subject><subject>Platelet Count</subject><subject>Portoenterostomy, Hepatic</subject><subject>Prognosis</subject><subject>Prospective Studies</subject><subject>Surgery</subject><issn>0022-3468</issn><issn>1531-5037</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2015</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNqFkU9v1DAQxS0EokvhK1Q5cknw2HFic0BUK_5JlTgUzpbjzEYO2XjxJJX22-NoWw5cOFljvTcz7zeM3QCvgEPzbqzGE_a0pqESHFTFZcU5PGM7UBJKxWX7nO04F6KUdaOv2CuikfP8zeEluxJKi7qWesfu9-clHnFwU3wIaaXSEUUf3IJ90YUpuHQu3JKQgntf3M6FwyXEKQ7Bu6lwc1-cUhzmSEvwBa3dkOJ6es1eHNxE-ObxvWY_P3_6sf9a3n3_8m1_e1f6Rpil9HmXVtetc1rJ_mA6Aa41QgB2Gg5aGACvnPICjG-gk6hzwY3yjTGi0Vxes7eXvnmH3yvSYo-BPE6TmzGuZKEVdU6qa5OlzUXqUyRKeLCnFI45nAVuN6B2tE9A7QbUcmkz0Gy8eZyxdkfs_9qeCGbBx4sAc9KHgMmSDzh77ENCv9g-hv_P-PBPCz-FeSP8C89IY1zTnDlasCQst_fbWbergsruRkr5B5LYn6M</recordid><startdate>20151001</startdate><enddate>20151001</enddate><creator>Zani, Augusto</creator><creator>Quaglia, Alberto</creator><creator>Hadzić, Nedim</creator><creator>Zuckerman, Mark</creator><creator>Davenport, Mark</creator><general>Elsevier Inc</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>7X8</scope><orcidid>https://orcid.org/0000-0002-5493-3418</orcidid></search><sort><creationdate>20151001</creationdate><title>Cytomegalovirus-associated biliary atresia: An aetiological and prognostic subgroup</title><author>Zani, Augusto ; Quaglia, Alberto ; Hadzić, Nedim ; Zuckerman, Mark ; Davenport, Mark</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c629t-c7017847aa853df9b21a79221eb81f82911c5a5c219c61b3e8a5c095c69926803</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2015</creationdate><topic>Age Factors</topic><topic>Aspartate Aminotransferases - blood</topic><topic>Biliary atresia</topic><topic>Biliary Atresia - pathology</topic><topic>Biliary Atresia - surgery</topic><topic>Biliary Atresia - virology</topic><topic>Cholestasis - etiology</topic><topic>Cytomegalovirus (CMV)</topic><topic>Cytomegalovirus - immunology</topic><topic>Cytomegalovirus Infections - complications</topic><topic>Female</topic><topic>Humans</topic><topic>Immunoglobulin M - analysis</topic><topic>Infant</topic><topic>Kasai portoenterostomy</topic><topic>Liver - enzymology</topic><topic>Liver - pathology</topic><topic>Liver - surgery</topic><topic>Male</topic><topic>Neonatal cholestatic jaundice</topic><topic>Pediatrics</topic><topic>Platelet Count</topic><topic>Portoenterostomy, Hepatic</topic><topic>Prognosis</topic><topic>Prospective Studies</topic><topic>Surgery</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Zani, Augusto</creatorcontrib><creatorcontrib>Quaglia, Alberto</creatorcontrib><creatorcontrib>Hadzić, Nedim</creatorcontrib><creatorcontrib>Zuckerman, Mark</creatorcontrib><creatorcontrib>Davenport, Mark</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>MEDLINE - Academic</collection><jtitle>Journal of pediatric surgery</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Zani, Augusto</au><au>Quaglia, Alberto</au><au>Hadzić, Nedim</au><au>Zuckerman, Mark</au><au>Davenport, Mark</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Cytomegalovirus-associated biliary atresia: An aetiological and prognostic subgroup</atitle><jtitle>Journal of pediatric surgery</jtitle><addtitle>J Pediatr Surg</addtitle><date>2015-10-01</date><risdate>2015</risdate><volume>50</volume><issue>10</issue><spage>1739</spage><epage>1745</epage><pages>1739-1745</pages><issn>0022-3468</issn><eissn>1531-5037</eissn><abstract>Abstract Background and aims Perinatal cytomegalovirus (CMV) infection is a possible cause or trigger of biliary atresia though clinical evidence is scant. We hypothesised that CMV IgM+ve biliary atresia is a separate clinical entity compared to CMV IgM−ve biliary atresia. Methods Prospective single-centre study. 210 infants with histologically confirmed biliary atresia were treated in our institution (Jan. 2004 to Dec. 2011); of these 20 (9.5%) were CMV IgM+ve at presentation. We compared these with 111 infants who were CMV IgM−ve (controls) for clinical features, biochemistry at presentation and outcome following Kasai portoenterostomy (KPE). A blinded comparison of age-matched liver histology was also performed. Data are quoted as median (interquartile range). A P value ≤ 0.05 was regarded as significant. Results Infants with CMV IgM+ve biliary atresia were older at Kasai portoenterostomy (or laparotomy) [70 (60–80) days vs. 56 (44–75)days; P = 0.003] and were more jaundiced [175 (147–224) vs. 140 (121–181) μmol/L; P = 0.002 + with higher AST*287 (157–403) vs. 180 (133–254) IU/L; P = 0.005] and aspartate aminotransferase-to-platelet ratio index [1.1 (0.79–3.0) vs. 0.63 (0.43–0.95)] levels. Liver histology : CMV IgM+ve biliary atresia was characterised by a greater degree of inflammation (P &lt; 0.0001) and fibrosis (P = 0.02), whereas CMV IgM−ve isolated biliary atresia had a higher degree of lobular cholestasis (P = 0.001). This effect was independent of the effects of age at KPE. Outcome : CMV IgM+ve biliary atresia had a poorer outcome with a reduced clearance of jaundice (15% vs. 52.2%; P = 0.002), native liver survival (P &lt; 0.0001) and increased mortality (P = 0.002). Conclusions CMV IgM+ve biliary atresia is a distinct clinical and pathological entity with a diminished response to Kasai portoenterostomy.</abstract><cop>United States</cop><pub>Elsevier Inc</pub><pmid>25824438</pmid><doi>10.1016/j.jpedsurg.2015.03.001</doi><tpages>7</tpages><orcidid>https://orcid.org/0000-0002-5493-3418</orcidid></addata></record>
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subjects Age Factors
Aspartate Aminotransferases - blood
Biliary atresia
Biliary Atresia - pathology
Biliary Atresia - surgery
Biliary Atresia - virology
Cholestasis - etiology
Cytomegalovirus (CMV)
Cytomegalovirus - immunology
Cytomegalovirus Infections - complications
Female
Humans
Immunoglobulin M - analysis
Infant
Kasai portoenterostomy
Liver - enzymology
Liver - pathology
Liver - surgery
Male
Neonatal cholestatic jaundice
Pediatrics
Platelet Count
Portoenterostomy, Hepatic
Prognosis
Prospective Studies
Surgery
title Cytomegalovirus-associated biliary atresia: An aetiological and prognostic subgroup
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