Survival of severe congenital diaphragmatic hernia has morbid consequences
Fetal tracheal occlusion (TO) was developed in an attempt to enhance prenatal lung growth and improve survival in fetuses with severe congenital diaphragmatic hernia (CDH). We conducted a randomized, controlled clinical trial in 24 fetuses with severe left CDH (liver herniated into the thorax and lo...
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| Veröffentlicht in: | Journal of pediatric surgery 2005, Vol.40 (1), p.36-46 |
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| creator | Cortes, Raul A. Keller, Roberta L. Townsend, Tiffany Harrison, Michael R. Farmer, Diana L. Lee, Hanmin Piecuch, Robert E. Leonard, Carol H. Hetherton, Maria Bisgaard, Robin Nobuhara, Kerilyn K. |
| description | Fetal tracheal occlusion (TO) was developed in an attempt to enhance prenatal lung growth and improve survival in fetuses with severe congenital diaphragmatic hernia (CDH). We conducted a randomized, controlled clinical trial in 24 fetuses with severe left CDH (liver herniated into the thorax and low lung-to-head ratio) to compare survival after endoscopic fetal TO vs standard perinatal care (control) and prospectively followed up the 16 survivors (9 control, 7 TO) to compare neurodevelopmental, respiratory, surgical, growth, and nutritional outcomes.
At 1 and 2 years old, subjects underwent evaluation consisting of medical and neurological history and physical, developmental testing, nutritional assessment, oxygen saturation and pulmonary function testing, chest radiograph, and echocardiogram. Growth and developmental measures were corrected for prematurity. Data were analyzed by Mann-Whitney rank sum test, Fisher's Exact test, and logistic and linear regression.
Infants with TO were significantly more premature at birth (control vs TO, 37.4 ± 1.0 vs 31.1 ± 1.7 weeks;
P < .01). Growth failure (
z score for weight |
| doi_str_mv | 10.1016/j.jpedsurg.2004.09.037 |
| format | Article |
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At 1 and 2 years old, subjects underwent evaluation consisting of medical and neurological history and physical, developmental testing, nutritional assessment, oxygen saturation and pulmonary function testing, chest radiograph, and echocardiogram. Growth and developmental measures were corrected for prematurity. Data were analyzed by Mann-Whitney rank sum test, Fisher's Exact test, and logistic and linear regression.
Infants with TO were significantly more premature at birth (control vs TO, 37.4 ± 1.0 vs 31.1 ± 1.7 weeks;
P < .01). Growth failure (
z score for weight <2 SDs below mean) was severe in both groups at 1 year of age (control vs TO, 56% vs 86%;
P = .31). There was considerable catch-up growth by age 2 years (growth failure: control vs TO, 22% vs 33%;
P = .19). There were no differences in other growth parameters. There were also no differences in neurodevelopmental outcome at 1 and 2 years. Supplemental oxygen at hospital discharge was a significant predictor of worse neurodevelopmental outcome at 1 and 2 years old (
P = .05 and
P = .02, respectively). Hearing loss requiring amplification has been diagnosed in 44% of the group (control vs TO, 44% vs 43%;
P = 1.0).
In this group of infants with severe CDH, there were no differences in outcome at 2 years old despite significant prematurity in the TO group. Oxygen supplementation at hospital discharge identified the most vulnerable group with respect to neurodevelopmental outcome, but all infants had significant growth failure, and hearing impairment is a substantial problem in this population. Severe CDH carries significant risk of chronic morbidity.</description><identifier>ISSN: 0022-3468</identifier><identifier>EISSN: 1531-5037</identifier><identifier>DOI: 10.1016/j.jpedsurg.2004.09.037</identifier><identifier>PMID: 15868556</identifier><language>eng</language><publisher>United States: Elsevier Inc</publisher><subject>Female ; Fetoscopy ; Follow-Up Studies ; Growth Disorders - etiology ; Hearing Disorders - etiology ; Hernia, Diaphragmatic - complications ; Hernia, Diaphragmatic - mortality ; Hernia, Diaphragmatic - surgery ; Humans ; Infant ; Infant, Newborn ; Infant, Premature ; Ligation - methods ; Lung - abnormalities ; Lung - embryology ; Lung Diseases - congenital ; Lung Diseases - etiology ; Lung Diseases - prevention & control ; Male ; Perinatal Care - methods ; Pregnancy ; Prospective Studies ; Trachea - surgery</subject><ispartof>Journal of pediatric surgery, 2005, Vol.40 (1), p.36-46</ispartof><rights>2005 Elsevier Inc.</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c366t-5105804f347f82aaf5a7a1f0a3e7dfc2c88af07930c5c095e0304b36b027019c3</citedby><cites>FETCH-LOGICAL-c366t-5105804f347f82aaf5a7a1f0a3e7dfc2c88af07930c5c095e0304b36b027019c3</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktohtml>$$Uhttps://dx.doi.org/10.1016/j.jpedsurg.2004.09.037$$EHTML$$P50$$Gelsevier$$H</linktohtml><link.rule.ids>314,776,780,3536,4009,27902,27903,27904,45974</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/15868556$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Cortes, Raul A.</creatorcontrib><creatorcontrib>Keller, Roberta L.</creatorcontrib><creatorcontrib>Townsend, Tiffany</creatorcontrib><creatorcontrib>Harrison, Michael R.</creatorcontrib><creatorcontrib>Farmer, Diana L.</creatorcontrib><creatorcontrib>Lee, Hanmin</creatorcontrib><creatorcontrib>Piecuch, Robert E.</creatorcontrib><creatorcontrib>Leonard, Carol H.</creatorcontrib><creatorcontrib>Hetherton, Maria</creatorcontrib><creatorcontrib>Bisgaard, Robin</creatorcontrib><creatorcontrib>Nobuhara, Kerilyn K.</creatorcontrib><title>Survival of severe congenital diaphragmatic hernia has morbid consequences</title><title>Journal of pediatric surgery</title><addtitle>J Pediatr Surg</addtitle><description>Fetal tracheal occlusion (TO) was developed in an attempt to enhance prenatal lung growth and improve survival in fetuses with severe congenital diaphragmatic hernia (CDH). We conducted a randomized, controlled clinical trial in 24 fetuses with severe left CDH (liver herniated into the thorax and low lung-to-head ratio) to compare survival after endoscopic fetal TO vs standard perinatal care (control) and prospectively followed up the 16 survivors (9 control, 7 TO) to compare neurodevelopmental, respiratory, surgical, growth, and nutritional outcomes.
At 1 and 2 years old, subjects underwent evaluation consisting of medical and neurological history and physical, developmental testing, nutritional assessment, oxygen saturation and pulmonary function testing, chest radiograph, and echocardiogram. Growth and developmental measures were corrected for prematurity. Data were analyzed by Mann-Whitney rank sum test, Fisher's Exact test, and logistic and linear regression.
Infants with TO were significantly more premature at birth (control vs TO, 37.4 ± 1.0 vs 31.1 ± 1.7 weeks;
P < .01). Growth failure (
z score for weight <2 SDs below mean) was severe in both groups at 1 year of age (control vs TO, 56% vs 86%;
P = .31). There was considerable catch-up growth by age 2 years (growth failure: control vs TO, 22% vs 33%;
P = .19). There were no differences in other growth parameters. There were also no differences in neurodevelopmental outcome at 1 and 2 years. Supplemental oxygen at hospital discharge was a significant predictor of worse neurodevelopmental outcome at 1 and 2 years old (
P = .05 and
P = .02, respectively). Hearing loss requiring amplification has been diagnosed in 44% of the group (control vs TO, 44% vs 43%;
P = 1.0).
In this group of infants with severe CDH, there were no differences in outcome at 2 years old despite significant prematurity in the TO group. Oxygen supplementation at hospital discharge identified the most vulnerable group with respect to neurodevelopmental outcome, but all infants had significant growth failure, and hearing impairment is a substantial problem in this population. Severe CDH carries significant risk of chronic morbidity.</description><subject>Female</subject><subject>Fetoscopy</subject><subject>Follow-Up Studies</subject><subject>Growth Disorders - etiology</subject><subject>Hearing Disorders - etiology</subject><subject>Hernia, Diaphragmatic - complications</subject><subject>Hernia, Diaphragmatic - mortality</subject><subject>Hernia, Diaphragmatic - surgery</subject><subject>Humans</subject><subject>Infant</subject><subject>Infant, Newborn</subject><subject>Infant, Premature</subject><subject>Ligation - methods</subject><subject>Lung - abnormalities</subject><subject>Lung - embryology</subject><subject>Lung Diseases - congenital</subject><subject>Lung Diseases - etiology</subject><subject>Lung Diseases - prevention & control</subject><subject>Male</subject><subject>Perinatal Care - methods</subject><subject>Pregnancy</subject><subject>Prospective Studies</subject><subject>Trachea - surgery</subject><issn>0022-3468</issn><issn>1531-5037</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2005</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNqFkMtOwzAQRS0EoqXwC1VW7BLGcewkOxDiqUosgLXlOOPWUZMUO6nE3-OoRSxZ2Rqd6zs-hCwpJBSouGmSZoe1H906SQGyBMoEWH5C5pQzGvNwPyVzgDSNWSaKGbnwvgEIY6DnZEZ5IQrOxZy8vo9ub_dqG_Um8rhHh5HuuzV2dgjD2qrdxql1qwarow26zqpoo3zU9q6y9YR6_Bqx0-gvyZlRW49Xx3NBPh8fPu6f49Xb08v93SrWTIgh5hR4AZlhWW6KVCnDVa6oAcUwr41OdVEoA3nJQHMNJUdgkFVMVJCG5UvNFuT68O7O9aHaD7K1XuN2qzrsRy9FHsIpFwEUB1C73nuHRu6cbZX7lhTkZFE28teinCxKKGVQFILLY8NYtVj_xY7aAnB7ADD8c2_RSa_tJKG2DvUg697-1_ED8J2Hiw</recordid><startdate>2005</startdate><enddate>2005</enddate><creator>Cortes, Raul A.</creator><creator>Keller, Roberta L.</creator><creator>Townsend, Tiffany</creator><creator>Harrison, Michael R.</creator><creator>Farmer, Diana L.</creator><creator>Lee, Hanmin</creator><creator>Piecuch, Robert E.</creator><creator>Leonard, Carol H.</creator><creator>Hetherton, Maria</creator><creator>Bisgaard, Robin</creator><creator>Nobuhara, Kerilyn K.</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></search><sort><creationdate>2005</creationdate><title>Survival of severe congenital diaphragmatic hernia has morbid consequences</title><author>Cortes, Raul A. ; Keller, Roberta L. ; Townsend, Tiffany ; Harrison, Michael R. ; Farmer, Diana L. ; Lee, Hanmin ; Piecuch, Robert E. ; Leonard, Carol H. ; Hetherton, Maria ; Bisgaard, Robin ; Nobuhara, Kerilyn K.</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c366t-5105804f347f82aaf5a7a1f0a3e7dfc2c88af07930c5c095e0304b36b027019c3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2005</creationdate><topic>Female</topic><topic>Fetoscopy</topic><topic>Follow-Up Studies</topic><topic>Growth Disorders - etiology</topic><topic>Hearing Disorders - etiology</topic><topic>Hernia, Diaphragmatic - complications</topic><topic>Hernia, Diaphragmatic - mortality</topic><topic>Hernia, Diaphragmatic - surgery</topic><topic>Humans</topic><topic>Infant</topic><topic>Infant, Newborn</topic><topic>Infant, Premature</topic><topic>Ligation - methods</topic><topic>Lung - abnormalities</topic><topic>Lung - embryology</topic><topic>Lung Diseases - congenital</topic><topic>Lung Diseases - etiology</topic><topic>Lung Diseases - prevention & control</topic><topic>Male</topic><topic>Perinatal Care - methods</topic><topic>Pregnancy</topic><topic>Prospective Studies</topic><topic>Trachea - surgery</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Cortes, Raul A.</creatorcontrib><creatorcontrib>Keller, Roberta L.</creatorcontrib><creatorcontrib>Townsend, Tiffany</creatorcontrib><creatorcontrib>Harrison, Michael R.</creatorcontrib><creatorcontrib>Farmer, Diana L.</creatorcontrib><creatorcontrib>Lee, Hanmin</creatorcontrib><creatorcontrib>Piecuch, Robert E.</creatorcontrib><creatorcontrib>Leonard, Carol H.</creatorcontrib><creatorcontrib>Hetherton, Maria</creatorcontrib><creatorcontrib>Bisgaard, Robin</creatorcontrib><creatorcontrib>Nobuhara, Kerilyn K.</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>Cortes, Raul A.</au><au>Keller, Roberta L.</au><au>Townsend, Tiffany</au><au>Harrison, Michael R.</au><au>Farmer, Diana L.</au><au>Lee, Hanmin</au><au>Piecuch, Robert E.</au><au>Leonard, Carol H.</au><au>Hetherton, Maria</au><au>Bisgaard, Robin</au><au>Nobuhara, Kerilyn K.</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Survival of severe congenital diaphragmatic hernia has morbid consequences</atitle><jtitle>Journal of pediatric surgery</jtitle><addtitle>J Pediatr Surg</addtitle><date>2005</date><risdate>2005</risdate><volume>40</volume><issue>1</issue><spage>36</spage><epage>46</epage><pages>36-46</pages><issn>0022-3468</issn><eissn>1531-5037</eissn><abstract>Fetal tracheal occlusion (TO) was developed in an attempt to enhance prenatal lung growth and improve survival in fetuses with severe congenital diaphragmatic hernia (CDH). We conducted a randomized, controlled clinical trial in 24 fetuses with severe left CDH (liver herniated into the thorax and low lung-to-head ratio) to compare survival after endoscopic fetal TO vs standard perinatal care (control) and prospectively followed up the 16 survivors (9 control, 7 TO) to compare neurodevelopmental, respiratory, surgical, growth, and nutritional outcomes.
At 1 and 2 years old, subjects underwent evaluation consisting of medical and neurological history and physical, developmental testing, nutritional assessment, oxygen saturation and pulmonary function testing, chest radiograph, and echocardiogram. Growth and developmental measures were corrected for prematurity. Data were analyzed by Mann-Whitney rank sum test, Fisher's Exact test, and logistic and linear regression.
Infants with TO were significantly more premature at birth (control vs TO, 37.4 ± 1.0 vs 31.1 ± 1.7 weeks;
P < .01). Growth failure (
z score for weight <2 SDs below mean) was severe in both groups at 1 year of age (control vs TO, 56% vs 86%;
P = .31). There was considerable catch-up growth by age 2 years (growth failure: control vs TO, 22% vs 33%;
P = .19). There were no differences in other growth parameters. There were also no differences in neurodevelopmental outcome at 1 and 2 years. Supplemental oxygen at hospital discharge was a significant predictor of worse neurodevelopmental outcome at 1 and 2 years old (
P = .05 and
P = .02, respectively). Hearing loss requiring amplification has been diagnosed in 44% of the group (control vs TO, 44% vs 43%;
P = 1.0).
In this group of infants with severe CDH, there were no differences in outcome at 2 years old despite significant prematurity in the TO group. Oxygen supplementation at hospital discharge identified the most vulnerable group with respect to neurodevelopmental outcome, but all infants had significant growth failure, and hearing impairment is a substantial problem in this population. Severe CDH carries significant risk of chronic morbidity.</abstract><cop>United States</cop><pub>Elsevier Inc</pub><pmid>15868556</pmid><doi>10.1016/j.jpedsurg.2004.09.037</doi><tpages>11</tpages></addata></record> |
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| ispartof | Journal of pediatric surgery, 2005, Vol.40 (1), p.36-46 |
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| language | eng |
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| source | MEDLINE; Elsevier ScienceDirect Journals |
| subjects | Female Fetoscopy Follow-Up Studies Growth Disorders - etiology Hearing Disorders - etiology Hernia, Diaphragmatic - complications Hernia, Diaphragmatic - mortality Hernia, Diaphragmatic - surgery Humans Infant Infant, Newborn Infant, Premature Ligation - methods Lung - abnormalities Lung - embryology Lung Diseases - congenital Lung Diseases - etiology Lung Diseases - prevention & control Male Perinatal Care - methods Pregnancy Prospective Studies Trachea - surgery |
| title | Survival of severe congenital diaphragmatic hernia has morbid consequences |
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