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Does neonatal screening of cystic fibrosis affect outcome? Comparative study of two cohorts in Britanny and Loire-Atlantique with follow-up after ten years
Neonatal screening for cystic fibrosis was started in Brittany in 1989 but not in the adjacent department of Loire-Atlantique. This study compares the outcome from the children of both populations nine years after the beginning of the screening. Those children were seen in different centers but with...
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| Published in: | Archives de pédiatrie : organe officiel de la Société française de pédiatrie 2000-11, Vol.7 (11), p.1154-1162 |
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| container_title | Archives de pédiatrie : organe officiel de la Société française de pédiatrie |
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| creator | Siret, D Branger, B Storni, V Bretaudeau, G Dagorne, M Moisan-Petit, V David, V Picherot, G Rault, G Roussey, M |
| description | Neonatal screening for cystic fibrosis was started in Brittany in 1989 but not in the adjacent department of Loire-Atlantique. This study compares the outcome from the children of both populations nine years after the beginning of the screening. Those children were seen in different centers but with the same following guidelines.
All children with cystic fibrosis born between 01/01/89 and 31/12/97 in Brittany and the Loire-Atlantique, excluding the meconium ileus, were compared for their initial characteristics and their outcome after nine years of follow-up.
There was no significant difference between both populations for sex ratio, gestational age, birth biometry, percentage of homozygotes delta F508, and mean age of children. Age at diagnosis was lower in Brittany (37 vs 372 days, P < 10(-7)), as was the delay for starting pancreatic supplementation (1.5 vs 14.3 months, P < 10(-7)). Percentage of children hospitalized at least once was higher in Loire-Atlantique (84.4 vs 40.3%, P < 10(-4)). There was no significant difference for colonization with Pseudomonas aeruginosa. Z-scores for weight and height were better in Brittany, as were Shwachman's and Brasfield's scores.
The homogeneity of both populations and their follow-up points out that even if the numbers of children are small and the study is retrospective, some benefits of neonatal screening appear, which are already found in other countries where it is partly practiced. This leads us recommend its general use in our populations, which should be associated with the follow-up of the screened children in cystic fibrosis centers to achieve the most of its benefits. |
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All children with cystic fibrosis born between 01/01/89 and 31/12/97 in Brittany and the Loire-Atlantique, excluding the meconium ileus, were compared for their initial characteristics and their outcome after nine years of follow-up.
There was no significant difference between both populations for sex ratio, gestational age, birth biometry, percentage of homozygotes delta F508, and mean age of children. Age at diagnosis was lower in Brittany (37 vs 372 days, P < 10(-7)), as was the delay for starting pancreatic supplementation (1.5 vs 14.3 months, P < 10(-7)). Percentage of children hospitalized at least once was higher in Loire-Atlantique (84.4 vs 40.3%, P < 10(-4)). There was no significant difference for colonization with Pseudomonas aeruginosa. Z-scores for weight and height were better in Brittany, as were Shwachman's and Brasfield's scores.
The homogeneity of both populations and their follow-up points out that even if the numbers of children are small and the study is retrospective, some benefits of neonatal screening appear, which are already found in other countries where it is partly practiced. This leads us recommend its general use in our populations, which should be associated with the follow-up of the screened children in cystic fibrosis centers to achieve the most of its benefits.</description><identifier>ISSN: 0929-693X</identifier><identifier>EISSN: 1769-664X</identifier><identifier>PMID: 11109941</identifier><language>fre</language><publisher>France</publisher><subject>Adolescent ; Child ; Child, Preschool ; Cohort Studies ; Cost-Benefit Analysis ; Cystic Fibrosis - diagnosis ; Cystic Fibrosis - pathology ; Female ; Humans ; Infant ; Infant, Newborn ; Infant, Newborn, Diseases - diagnosis ; Male ; Neonatal Screening ; Outcome Assessment (Health Care) ; Severity of Illness Index</subject><ispartof>Archives de pédiatrie : organe officiel de la Société française de pédiatrie, 2000-11, Vol.7 (11), p.1154-1162</ispartof><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>315,786,790</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/11109941$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Siret, D</creatorcontrib><creatorcontrib>Branger, B</creatorcontrib><creatorcontrib>Storni, V</creatorcontrib><creatorcontrib>Bretaudeau, G</creatorcontrib><creatorcontrib>Dagorne, M</creatorcontrib><creatorcontrib>Moisan-Petit, V</creatorcontrib><creatorcontrib>David, V</creatorcontrib><creatorcontrib>Picherot, G</creatorcontrib><creatorcontrib>Rault, G</creatorcontrib><creatorcontrib>Roussey, M</creatorcontrib><title>Does neonatal screening of cystic fibrosis affect outcome? Comparative study of two cohorts in Britanny and Loire-Atlantique with follow-up after ten years</title><title>Archives de pédiatrie : organe officiel de la Société française de pédiatrie</title><addtitle>Arch Pediatr</addtitle><description>Neonatal screening for cystic fibrosis was started in Brittany in 1989 but not in the adjacent department of Loire-Atlantique. This study compares the outcome from the children of both populations nine years after the beginning of the screening. Those children were seen in different centers but with the same following guidelines.
All children with cystic fibrosis born between 01/01/89 and 31/12/97 in Brittany and the Loire-Atlantique, excluding the meconium ileus, were compared for their initial characteristics and their outcome after nine years of follow-up.
There was no significant difference between both populations for sex ratio, gestational age, birth biometry, percentage of homozygotes delta F508, and mean age of children. Age at diagnosis was lower in Brittany (37 vs 372 days, P < 10(-7)), as was the delay for starting pancreatic supplementation (1.5 vs 14.3 months, P < 10(-7)). Percentage of children hospitalized at least once was higher in Loire-Atlantique (84.4 vs 40.3%, P < 10(-4)). There was no significant difference for colonization with Pseudomonas aeruginosa. Z-scores for weight and height were better in Brittany, as were Shwachman's and Brasfield's scores.
The homogeneity of both populations and their follow-up points out that even if the numbers of children are small and the study is retrospective, some benefits of neonatal screening appear, which are already found in other countries where it is partly practiced. This leads us recommend its general use in our populations, which should be associated with the follow-up of the screened children in cystic fibrosis centers to achieve the most of its benefits.</description><subject>Adolescent</subject><subject>Child</subject><subject>Child, Preschool</subject><subject>Cohort Studies</subject><subject>Cost-Benefit Analysis</subject><subject>Cystic Fibrosis - diagnosis</subject><subject>Cystic Fibrosis - pathology</subject><subject>Female</subject><subject>Humans</subject><subject>Infant</subject><subject>Infant, Newborn</subject><subject>Infant, Newborn, Diseases - diagnosis</subject><subject>Male</subject><subject>Neonatal Screening</subject><subject>Outcome Assessment (Health Care)</subject><subject>Severity of Illness Index</subject><issn>0929-693X</issn><issn>1769-664X</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2000</creationdate><recordtype>article</recordtype><recordid>eNo1kMtOwzAQRS0EoqXwC2hW7CLZeTlZoVKeUiU2XXQXOc6YGiV2sB2qfAs_SyLKau5Id-6cmTOyZDwvozxP9-dkSct40mWyX5Ar7z8ppQUtkkuyYIzRskzZkvw8WvRg0BoRRAteOkSjzQdYBXL0QUtQunbWaw9CKZQB7BCk7fAeNrbrhRNBfyP4MDTjPBSOFqQ9WBc8aAMPTgdhzAjCNLC12mG0Dq0wQX8NCEcdDqBs29pjNPTTgoAOAhoYUTh_TS6UaD3enOqK7J6fdpvXaPv-8rZZb6M-S1lUp4zlmUSu2HQdVQVtZk7Bp4Y3MRM8liLjmHDeKBQ0Y7FiitcUJUdWx8mK3P3F9s5OUD5UnfYS24kS7eArHqeclsVsvD0Zh7rDpuqd7oQbq_9vJr9vwXYC</recordid><startdate>200011</startdate><enddate>200011</enddate><creator>Siret, D</creator><creator>Branger, B</creator><creator>Storni, V</creator><creator>Bretaudeau, G</creator><creator>Dagorne, M</creator><creator>Moisan-Petit, V</creator><creator>David, V</creator><creator>Picherot, G</creator><creator>Rault, G</creator><creator>Roussey, M</creator><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>7X8</scope></search><sort><creationdate>200011</creationdate><title>Does neonatal screening of cystic fibrosis affect outcome? Comparative study of two cohorts in Britanny and Loire-Atlantique with follow-up after ten years</title><author>Siret, D ; Branger, B ; Storni, V ; Bretaudeau, G ; Dagorne, M ; Moisan-Petit, V ; David, V ; Picherot, G ; Rault, G ; Roussey, M</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-p541-b41165ce7f18080f80dffeca70807d21a72ca57e377dfea0512f1f7b0ec7e1b23</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>fre</language><creationdate>2000</creationdate><topic>Adolescent</topic><topic>Child</topic><topic>Child, Preschool</topic><topic>Cohort Studies</topic><topic>Cost-Benefit Analysis</topic><topic>Cystic Fibrosis - diagnosis</topic><topic>Cystic Fibrosis - pathology</topic><topic>Female</topic><topic>Humans</topic><topic>Infant</topic><topic>Infant, Newborn</topic><topic>Infant, Newborn, Diseases - diagnosis</topic><topic>Male</topic><topic>Neonatal Screening</topic><topic>Outcome Assessment (Health Care)</topic><topic>Severity of Illness Index</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Siret, D</creatorcontrib><creatorcontrib>Branger, B</creatorcontrib><creatorcontrib>Storni, V</creatorcontrib><creatorcontrib>Bretaudeau, G</creatorcontrib><creatorcontrib>Dagorne, M</creatorcontrib><creatorcontrib>Moisan-Petit, V</creatorcontrib><creatorcontrib>David, V</creatorcontrib><creatorcontrib>Picherot, G</creatorcontrib><creatorcontrib>Rault, G</creatorcontrib><creatorcontrib>Roussey, M</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>MEDLINE - Academic</collection><jtitle>Archives de pédiatrie : organe officiel de la Société française de pédiatrie</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Siret, D</au><au>Branger, B</au><au>Storni, V</au><au>Bretaudeau, G</au><au>Dagorne, M</au><au>Moisan-Petit, V</au><au>David, V</au><au>Picherot, G</au><au>Rault, G</au><au>Roussey, M</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Does neonatal screening of cystic fibrosis affect outcome? Comparative study of two cohorts in Britanny and Loire-Atlantique with follow-up after ten years</atitle><jtitle>Archives de pédiatrie : organe officiel de la Société française de pédiatrie</jtitle><addtitle>Arch Pediatr</addtitle><date>2000-11</date><risdate>2000</risdate><volume>7</volume><issue>11</issue><spage>1154</spage><epage>1162</epage><pages>1154-1162</pages><issn>0929-693X</issn><eissn>1769-664X</eissn><notes>ObjectType-Article-2</notes><notes>SourceType-Scholarly Journals-1</notes><notes>ObjectType-Feature-1</notes><notes>content type line 23</notes><abstract>Neonatal screening for cystic fibrosis was started in Brittany in 1989 but not in the adjacent department of Loire-Atlantique. This study compares the outcome from the children of both populations nine years after the beginning of the screening. Those children were seen in different centers but with the same following guidelines.
All children with cystic fibrosis born between 01/01/89 and 31/12/97 in Brittany and the Loire-Atlantique, excluding the meconium ileus, were compared for their initial characteristics and their outcome after nine years of follow-up.
There was no significant difference between both populations for sex ratio, gestational age, birth biometry, percentage of homozygotes delta F508, and mean age of children. Age at diagnosis was lower in Brittany (37 vs 372 days, P < 10(-7)), as was the delay for starting pancreatic supplementation (1.5 vs 14.3 months, P < 10(-7)). Percentage of children hospitalized at least once was higher in Loire-Atlantique (84.4 vs 40.3%, P < 10(-4)). There was no significant difference for colonization with Pseudomonas aeruginosa. Z-scores for weight and height were better in Brittany, as were Shwachman's and Brasfield's scores.
The homogeneity of both populations and their follow-up points out that even if the numbers of children are small and the study is retrospective, some benefits of neonatal screening appear, which are already found in other countries where it is partly practiced. This leads us recommend its general use in our populations, which should be associated with the follow-up of the screened children in cystic fibrosis centers to achieve the most of its benefits.</abstract><cop>France</cop><pmid>11109941</pmid><tpages>9</tpages></addata></record> |
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| identifier | ISSN: 0929-693X |
| ispartof | Archives de pédiatrie : organe officiel de la Société française de pédiatrie, 2000-11, Vol.7 (11), p.1154-1162 |
| issn | 0929-693X 1769-664X |
| language | fre |
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| source | ScienceDirect Freedom Collection |
| subjects | Adolescent Child Child, Preschool Cohort Studies Cost-Benefit Analysis Cystic Fibrosis - diagnosis Cystic Fibrosis - pathology Female Humans Infant Infant, Newborn Infant, Newborn, Diseases - diagnosis Male Neonatal Screening Outcome Assessment (Health Care) Severity of Illness Index |
| title | Does neonatal screening of cystic fibrosis affect outcome? Comparative study of two cohorts in Britanny and Loire-Atlantique with follow-up after ten years |
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