A proposed mechanism for intermediate atresia ani (AA), based on a porcine case of AA and hypospadias
BACKGROUND Atresia Ani (AA) is one of the most common malformations of the lower gastrointestinal tract. It occurs in 1 in 1500 to 1 in 5000 human births. These patients suffer physically and psychologically from this disorder, and thus there is a research interest in the condition. There are 3 subc...
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Veröffentlicht in: | Birth defects research. A Clinical and molecular teratology 2005-06, Vol.73 (6), p.434-439 |
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creator | Finnigan, Daniel F. Fisher, Ken R.S. Vrablic, Olga Halina, Winn G. Partlow, Gary D. |
description | BACKGROUND
Atresia Ani (AA) is one of the most common malformations of the lower gastrointestinal tract. It occurs in 1 in 1500 to 1 in 5000 human births. These patients suffer physically and psychologically from this disorder, and thus there is a research interest in the condition. There are 3 subcategories of AA: high, intermediate, and low. Each of these forms is likely related to separate etiological processes.
METHODS
An anatomical study was performed on a porcine case of AA with a urorectal fistula and malformed urethra.
RESULTS
This animal was found to have the intermediate form of AA.
CONCLUSIONS
A new mechanism is hypothesized, distinct from that described for the high and low forms of AA. This proposed mechanism involves the persistence of the cloacal membrane. Evidence to support this hypothesis includes: location of the urorectal fistula in the region of the embryonic cloacal duct, the lack of anomalies usually seen in conjunction with AA associated with mutations of the Sonic Hedgehog gene, and the presence of a malformed urethra, which is significantly correlated to intermediate AA. This form of AA could be related to a failure of the cloacal membrane to break down at the appropriate time during urorectal separation. Birth Defects Research (Part A), 2005. © 2005 Wiley‐Liss, Inc. |
doi_str_mv | 10.1002/bdra.20129 |
format | Article |
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Atresia Ani (AA) is one of the most common malformations of the lower gastrointestinal tract. It occurs in 1 in 1500 to 1 in 5000 human births. These patients suffer physically and psychologically from this disorder, and thus there is a research interest in the condition. There are 3 subcategories of AA: high, intermediate, and low. Each of these forms is likely related to separate etiological processes.
METHODS
An anatomical study was performed on a porcine case of AA with a urorectal fistula and malformed urethra.
RESULTS
This animal was found to have the intermediate form of AA.
CONCLUSIONS
A new mechanism is hypothesized, distinct from that described for the high and low forms of AA. This proposed mechanism involves the persistence of the cloacal membrane. Evidence to support this hypothesis includes: location of the urorectal fistula in the region of the embryonic cloacal duct, the lack of anomalies usually seen in conjunction with AA associated with mutations of the Sonic Hedgehog gene, and the presence of a malformed urethra, which is significantly correlated to intermediate AA. This form of AA could be related to a failure of the cloacal membrane to break down at the appropriate time during urorectal separation. Birth Defects Research (Part A), 2005. © 2005 Wiley‐Liss, Inc.</description><identifier>ISSN: 1542-0752</identifier><identifier>EISSN: 1542-0760</identifier><identifier>DOI: 10.1002/bdra.20129</identifier><identifier>PMID: 15880702</identifier><identifier>CODEN: BDRPBT</identifier><language>eng</language><publisher>Hoboken: Wiley Subscription Services, Inc., A Wiley Company</publisher><subject>Animals ; Anus, Imperforate - etiology ; Anus, Imperforate - pathology ; atresia ani ; Biological and medical sciences ; Disease Models, Animal ; Embryology: invertebrates and vertebrates. Teratology ; Fundamental and applied biological sciences. Psychology ; gastrointestinal malformation ; hypospadias ; Hypospadias - etiology ; Hypospadias - pathology ; Male ; Swine ; Teratology. Teratogens ; urethral malformation ; urogenital malformation ; urorectal malformation</subject><ispartof>Birth defects research. A Clinical and molecular teratology, 2005-06, Vol.73 (6), p.434-439</ispartof><rights>Copyright © 2005 Wiley‐Liss, Inc.</rights><rights>2005 INIST-CNRS</rights><rights>(c) 2005 Wiley-Liss, Inc.</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><cites>FETCH-LOGICAL-c3549-6fa9f5b12927a56257cb43da744c5120e7df766e590889eafac99ec2ebef92263</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://onlinelibrary.wiley.com/doi/pdf/10.1002%2Fbdra.20129$$EPDF$$P50$$Gwiley$$H</linktopdf><linktohtml>$$Uhttps://onlinelibrary.wiley.com/doi/full/10.1002%2Fbdra.20129$$EHTML$$P50$$Gwiley$$H</linktohtml><link.rule.ids>314,777,781,1412,27905,27906,45555,45556</link.rule.ids><backlink>$$Uhttp://pascal-francis.inist.fr/vibad/index.php?action=getRecordDetail&idt=16877953$$DView record in Pascal Francis$$Hfree_for_read</backlink><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/15880702$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Finnigan, Daniel F.</creatorcontrib><creatorcontrib>Fisher, Ken R.S.</creatorcontrib><creatorcontrib>Vrablic, Olga</creatorcontrib><creatorcontrib>Halina, Winn G.</creatorcontrib><creatorcontrib>Partlow, Gary D.</creatorcontrib><title>A proposed mechanism for intermediate atresia ani (AA), based on a porcine case of AA and hypospadias</title><title>Birth defects research. A Clinical and molecular teratology</title><addtitle>Birth Defects Research Part A: Clinical and Molecular Teratology</addtitle><description>BACKGROUND
Atresia Ani (AA) is one of the most common malformations of the lower gastrointestinal tract. It occurs in 1 in 1500 to 1 in 5000 human births. These patients suffer physically and psychologically from this disorder, and thus there is a research interest in the condition. There are 3 subcategories of AA: high, intermediate, and low. Each of these forms is likely related to separate etiological processes.
METHODS
An anatomical study was performed on a porcine case of AA with a urorectal fistula and malformed urethra.
RESULTS
This animal was found to have the intermediate form of AA.
CONCLUSIONS
A new mechanism is hypothesized, distinct from that described for the high and low forms of AA. This proposed mechanism involves the persistence of the cloacal membrane. Evidence to support this hypothesis includes: location of the urorectal fistula in the region of the embryonic cloacal duct, the lack of anomalies usually seen in conjunction with AA associated with mutations of the Sonic Hedgehog gene, and the presence of a malformed urethra, which is significantly correlated to intermediate AA. This form of AA could be related to a failure of the cloacal membrane to break down at the appropriate time during urorectal separation. Birth Defects Research (Part A), 2005. © 2005 Wiley‐Liss, Inc.</description><subject>Animals</subject><subject>Anus, Imperforate - etiology</subject><subject>Anus, Imperforate - pathology</subject><subject>atresia ani</subject><subject>Biological and medical sciences</subject><subject>Disease Models, Animal</subject><subject>Embryology: invertebrates and vertebrates. Teratology</subject><subject>Fundamental and applied biological sciences. Psychology</subject><subject>gastrointestinal malformation</subject><subject>hypospadias</subject><subject>Hypospadias - etiology</subject><subject>Hypospadias - pathology</subject><subject>Male</subject><subject>Swine</subject><subject>Teratology. Teratogens</subject><subject>urethral malformation</subject><subject>urogenital malformation</subject><subject>urorectal malformation</subject><issn>1542-0752</issn><issn>1542-0760</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2005</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNp9kEtv1DAUhS1ERV9s-AHIG1CpSOs4sR0v01JaRlUrVSCW1o1zrRryws6Izr_H05m2O1bXuv7OuTqHkHc5O8kZ46dNG-CEs5zrV2QvFyXPmJLs9fNb8F2yH-OvxBZKqTdkNxdVxRTjewRrOoVxGiO2tEd7D4OPPXVjoH6YMfTYepiRwhwweqDpmx7V9afPtIG1ZBwo0GkM1g9IbVrR0dG6TlxL71fJdoJkEA_JjoMu4tvtPCA_vl58P7_Krm8vv53X15ktRKkz6UA70aQgXIGQXCjblEULqiytyDlD1TolJQrNqkojOLBao-XYoNOcy-KAfNz4pkx_lhhn0_tosetgwHEZjVSaF0yvweMNaMMYY0BnpuB7CCuTM7Mu1axLNY-lJvj91nXZpEJe0G2LCfiwBSBa6FyAwfr4wslKKS2KxOUb7q_vcPWfk-bsy139dDzbaHyc8eFZA-F3SlMoYX7eXBrJ2ILdLbhZFP8A19mcXw</recordid><startdate>200506</startdate><enddate>200506</enddate><creator>Finnigan, Daniel F.</creator><creator>Fisher, Ken R.S.</creator><creator>Vrablic, Olga</creator><creator>Halina, Winn G.</creator><creator>Partlow, Gary D.</creator><general>Wiley Subscription Services, Inc., A Wiley Company</general><general>Wiley</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>7X8</scope></search><sort><creationdate>200506</creationdate><title>A proposed mechanism for intermediate atresia ani (AA), based on a porcine case of AA and hypospadias</title><author>Finnigan, Daniel F. ; Fisher, Ken R.S. ; Vrablic, Olga ; Halina, Winn G. ; Partlow, Gary D.</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c3549-6fa9f5b12927a56257cb43da744c5120e7df766e590889eafac99ec2ebef92263</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2005</creationdate><topic>Animals</topic><topic>Anus, Imperforate - etiology</topic><topic>Anus, Imperforate - pathology</topic><topic>atresia ani</topic><topic>Biological and medical sciences</topic><topic>Disease Models, Animal</topic><topic>Embryology: invertebrates and vertebrates. Teratology</topic><topic>Fundamental and applied biological sciences. Psychology</topic><topic>gastrointestinal malformation</topic><topic>hypospadias</topic><topic>Hypospadias - etiology</topic><topic>Hypospadias - pathology</topic><topic>Male</topic><topic>Swine</topic><topic>Teratology. Teratogens</topic><topic>urethral malformation</topic><topic>urogenital malformation</topic><topic>urorectal malformation</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Finnigan, Daniel F.</creatorcontrib><creatorcontrib>Fisher, Ken R.S.</creatorcontrib><creatorcontrib>Vrablic, Olga</creatorcontrib><creatorcontrib>Halina, Winn G.</creatorcontrib><creatorcontrib>Partlow, Gary D.</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>MEDLINE - Academic</collection><jtitle>Birth defects research. A Clinical and molecular teratology</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Finnigan, Daniel F.</au><au>Fisher, Ken R.S.</au><au>Vrablic, Olga</au><au>Halina, Winn G.</au><au>Partlow, Gary D.</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>A proposed mechanism for intermediate atresia ani (AA), based on a porcine case of AA and hypospadias</atitle><jtitle>Birth defects research. A Clinical and molecular teratology</jtitle><addtitle>Birth Defects Research Part A: Clinical and Molecular Teratology</addtitle><date>2005-06</date><risdate>2005</risdate><volume>73</volume><issue>6</issue><spage>434</spage><epage>439</epage><pages>434-439</pages><issn>1542-0752</issn><eissn>1542-0760</eissn><coden>BDRPBT</coden><abstract>BACKGROUND
Atresia Ani (AA) is one of the most common malformations of the lower gastrointestinal tract. It occurs in 1 in 1500 to 1 in 5000 human births. These patients suffer physically and psychologically from this disorder, and thus there is a research interest in the condition. There are 3 subcategories of AA: high, intermediate, and low. Each of these forms is likely related to separate etiological processes.
METHODS
An anatomical study was performed on a porcine case of AA with a urorectal fistula and malformed urethra.
RESULTS
This animal was found to have the intermediate form of AA.
CONCLUSIONS
A new mechanism is hypothesized, distinct from that described for the high and low forms of AA. This proposed mechanism involves the persistence of the cloacal membrane. Evidence to support this hypothesis includes: location of the urorectal fistula in the region of the embryonic cloacal duct, the lack of anomalies usually seen in conjunction with AA associated with mutations of the Sonic Hedgehog gene, and the presence of a malformed urethra, which is significantly correlated to intermediate AA. This form of AA could be related to a failure of the cloacal membrane to break down at the appropriate time during urorectal separation. Birth Defects Research (Part A), 2005. © 2005 Wiley‐Liss, Inc.</abstract><cop>Hoboken</cop><pub>Wiley Subscription Services, Inc., A Wiley Company</pub><pmid>15880702</pmid><doi>10.1002/bdra.20129</doi><tpages>6</tpages></addata></record> |
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subjects | Animals Anus, Imperforate - etiology Anus, Imperforate - pathology atresia ani Biological and medical sciences Disease Models, Animal Embryology: invertebrates and vertebrates. Teratology Fundamental and applied biological sciences. Psychology gastrointestinal malformation hypospadias Hypospadias - etiology Hypospadias - pathology Male Swine Teratology. Teratogens urethral malformation urogenital malformation urorectal malformation |
title | A proposed mechanism for intermediate atresia ani (AA), based on a porcine case of AA and hypospadias |
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