{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,6]],"date-time":"2026-10-06T08:22:46Z","timestamp":1791274966411,"version":"4.1.0"},"reference-count":41,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2001,3,21]],"date-time":"2001-03-21T00:00:00Z","timestamp":985132800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Anat. Rec."],"published-print":{"date-parts":[[2001,5]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>The <jats:italic>inv\/inv<\/jats:italic> mouse carries an insertional mutation in the inversin gene, (<jats:italic>inv<\/jats:italic>, for inversion of embryonic turning). Previously it had been reported that almost 100% of the homozygous offspring (<jats:italic>inv\/inv<\/jats:italic>) were characterized by situs inversus totalis. In this report we identify the spectrum of cardiopulmonary anatomical abnormalities in <jats:italic>inv\/inv<\/jats:italic> mice surviving to birth to determine whether the abnormalities seen are of the categories classically associated with human situs abnormalities. Stillborn mice, offspring that died unexpectedly (within 48 hr after birth), and neonates with phenotypic characteristics of situs inversus (right\u2010sided stomachs, growth failure or jaundice) were processed for standard histological examination. Of 173 offspring, 34 (20%) neonates (11 stillborn, 9 unexpected deaths, and 14 mice with situs inversus phenotype) were examined, 27 of which were genotyped to be <jats:italic>inv\/inv<\/jats:italic>. Interestingly, three <jats:italic>inv\/inv<\/jats:italic> mice (11%) were found to have situs solitus. Twenty\u2010four had situs inversus with normal, mirror\u2010image cardiac anatomy (dextrocardia with atrioventricular concordance, ventriculoarterial concordance and a right aortic arch). The overall incidence of cardiovascular anomalies observed was 10 out of 27 (37%). The most frequent severe malformation, identified in 3 out of 27 animals, was a complex consisting of pulmonary infundibular stenosis\/atresia with absence of pulmonary valve tissue and a ventricular septal defect. The pulmonary phenotype in <jats:italic>inv\/inv<\/jats:italic> mice was situs inversus with occasional minor lobar abnormalities. We conclude that 1) cardiopulmonary malformations in <jats:italic>inv\/inv<\/jats:italic> mice are not rare (37%), 2) the cardiopulmonary malformations observed in <jats:italic>inv\/inv<\/jats:italic> specimens are not of the spectrum typically associated with human heterotaxia. In particular, <jats:italic>inv\/inv<\/jats:italic> mice have a propensity for defects in the development of the right ventricular outflow tract and the interventricular septum, and 3) approximately one out of ten <jats:italic>inv\/inv<\/jats:italic> mice is born with situs solitus and shows cardiac anomalies that correspond to those observed in <jats:italic>inv\/inv<\/jats:italic> specimens with situs inversus. Our data therefore suggest that inversin, the product of the <jats:italic>inv<\/jats:italic> locus, may have specific roles in cardiac morphogenesis independent of its role in situs determination. Anat Rec 263:62\u201371, 2001. \u00a9 2001 Wiley\u2010Liss, Inc.<\/jats:p>","DOI":"10.1002\/ar.1077","type":"journal-article","created":{"date-parts":[[2002,8,25]],"date-time":"2002-08-25T20:21:39Z","timestamp":1030306899000},"page":"62-71","source":"Crossref","is-referenced-by-count":25,"title":["Cardiopulmonary malformations in the <i>inv\/inv<\/i> mouse"],"prefix":"10.1002","volume":"263","author":[{"given":"Tim C.","family":"McQuinn","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Daniel E.","family":"Miga","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Corey H.","family":"Mjaatvedt","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aimee L.","family":"Phelps","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andy","family":"Wessels","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2001,3,21]]},"reference":[{"key":"e_1_2_6_2_1","first-page":"852","article-title":"Genetical and ultrastructural aspects of the immotile\u2010cilia syndrome","volume":"33","author":"Afzelius BA","year":"1981","journal-title":"Am J Hum Genet"},{"key":"e_1_2_6_3_1","doi-asserted-by":"crossref","first-page":"658","DOI":"10.1038\/35001072","article-title":"The organizer factors Chordin and Noggin are required for mouse forebrain development","volume":"402","author":"Bachiller D","year":"2000","journal-title":"Nature"},{"key":"e_1_2_6_4_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.126.6.1225"},{"key":"e_1_2_6_5_1","doi-asserted-by":"crossref","first-page":"1631","DOI":"10.1242\/dev.126.8.1631","article-title":"Smad5 knockout mice die at mid\u2010gestation due to multiple embryonic and extraembryonic defect","volume":"126","author":"Chang H","year":"1999","journal-title":"Development"},{"key":"e_1_2_6_6_1","doi-asserted-by":"publisher","DOI":"10.1006\/dbio.1999.9594"},{"key":"e_1_2_6_7_1","doi-asserted-by":"crossref","first-page":"3015","DOI":"10.1242\/dev.125.16.3015","article-title":"The transcription factor HNF3\u03b2 is required in visceral endoderm for normal primitive streak morphogenesis","volume":"125","author":"Dufort D","year":"1998","journal-title":"Development"},{"key":"e_1_2_6_8_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng1197-305"},{"key":"e_1_2_6_9_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0022-3476(76)80945-6"},{"key":"e_1_2_6_10_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.96.22.12595"},{"key":"e_1_2_6_11_1","doi-asserted-by":"publisher","DOI":"10.1161\/01.CIR.84.6.2547"},{"key":"e_1_2_6_12_1","doi-asserted-by":"publisher","DOI":"10.1038\/21429"},{"key":"e_1_2_6_13_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0925-4773(98)00166-X"},{"key":"e_1_2_6_14_1","doi-asserted-by":"publisher","DOI":"10.1016\/B978-012329860-7\/50013-1"},{"key":"e_1_2_6_15_1","doi-asserted-by":"publisher","DOI":"10.1093\/oxfordjournals.jhered.a108749"},{"key":"e_1_2_6_16_1","doi-asserted-by":"publisher","DOI":"10.1161\/01.CIR.96.3.874"},{"key":"e_1_2_6_17_1","doi-asserted-by":"publisher","DOI":"10.1038\/45803"},{"key":"e_1_2_6_18_1","doi-asserted-by":"publisher","DOI":"10.1038\/45797"},{"key":"e_1_2_6_19_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.96.9.5043"},{"key":"e_1_2_6_20_1","doi-asserted-by":"publisher","DOI":"10.1002\/hep.510300223"},{"key":"e_1_2_6_21_1","unstructured":"McKusickV. 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