{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,5]],"date-time":"2026-10-05T10:35:22Z","timestamp":1791196522327,"version":"4.1.0"},"reference-count":56,"publisher":"The Company of Biologists","issue":"23","license":[{"start":{"date-parts":[[1997,12,1]],"date-time":"1997-12-01T00:00:00Z","timestamp":880934400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/www.biologists.com\/user-licence-1-1\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[1997,12,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n               <jats:p>Gene targeting experiments have shown that the murine Hoxa-13 and Hoxd-13 paralogous genes control skeletal patterning in the distal region of the developing limbs. However, both genes are also expressed in the terminal part of the digestive and urogenital tracts during embryogenesis and postnatal development. Here, we report the abnormalities occuring in these systems in Hoxa-13\u2212\/\u2212 and Hoxa-13\/Hoxd-13 compound mutant mice. Hoxa-13\u2212\/\u2212 mutant fetuses show agenesis of the caudal portion of the M\u00fcllerian ducts, lack of development of the presumptive urinary bladder and premature stenosis of the umbilical arteries, which could account for the lethality of this mutation at mid-gestational stages. Due to such lethality, only Hoxa-13+\/\u2212\/Hoxd-13\u2212\/\u2212 compound mutants can reach adulthood. These compound mutants display: (i) agenesis or hypoplasia of some of the male accessory sex glands, (ii) malpositioning of the vaginal, urethral and anal openings, and improper separation of the vagina from the urogenital sinus, (iii) hydronephrosis and (iv) anomalies of the muscular and epithelial layers of the rectum. Thus, Hoxa-13 and Hoxd-13 play important roles in the morphogenesis of the terminal part of the gut and urogenital tract. While Hoxa-13\u2212\/\u2212\/Hoxd-13+\/\u2212 fetuses show severely impaired development of the urogenital sinus, double null (Hoxa-13\u2212\/\u2212\/Hoxd-13\u2212\/\u2212) fetuses display no separation of the terminal (cloacal) hindgut cavity into a urogenital sinus and presumptive rectum, and no development of the genital bud, thereby demonstrating that both genes act, in a partly redundant manner, during early morphogenesis of posterior trunk structures.<\/jats:p>","DOI":"10.1242\/dev.124.23.4781","type":"journal-article","created":{"date-parts":[[2021,4,26]],"date-time":"2021-04-26T03:10:20Z","timestamp":1619406620000},"page":"4781-4791","source":"Crossref","is-referenced-by-count":316,"title":["Gene dosage-dependent effects of the <i>Hoxa-13<\/i> and <i>Hoxd-13<\/i> mutations on morphogenesis of the terminal parts of the digestive and urogenital tracts"],"prefix":"10.1242","volume":"124","author":[{"given":"Xavier","family":"Warot","sequence":"first","affiliation":[{"name":"Institut de G\u00e9n\u00e9tique et de Biologie Mol\u00e9culaire et Cellulaire, CNRS\/INSERM\/ULP\/Coll\u00e8ge de France , BP 163, 67404 ILLKIRCH Cedex, CU de Strasbourg, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Catherine","family":"Fromental-Ramain","sequence":"additional","affiliation":[{"name":"Institut de G\u00e9n\u00e9tique et de Biologie Mol\u00e9culaire et Cellulaire, CNRS\/INSERM\/ULP\/Coll\u00e8ge de France , BP 163, 67404 ILLKIRCH Cedex, CU de Strasbourg, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Val\u00e9rie","family":"Fraulob","sequence":"additional","affiliation":[{"name":"Institut de G\u00e9n\u00e9tique et de Biologie Mol\u00e9culaire et Cellulaire, CNRS\/INSERM\/ULP\/Coll\u00e8ge de France , BP 163, 67404 ILLKIRCH Cedex, CU de Strasbourg, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pierre","family":"Chambon","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pascal","family":"Doll\u00e9","sequence":"additional","affiliation":[{"name":"Institut de G\u00e9n\u00e9tique et de Biologie Mol\u00e9culaire et Cellulaire, CNRS\/INSERM\/ULP\/Coll\u00e8ge de France , BP 163, 67404 ILLKIRCH Cedex, CU de Strasbourg, France"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[1997,12,1]]},"reference":[{"key":"2025021406042414200_DEVELOP_124_23_4781C1","doi-asserted-by":"publisher","first-page":"2687","DOI":"10.1242\/dev.122.9.2687","article-title":"Mechanisms of reduced fertility in Hoxa-10 mutant mice: uterine homeosis and loss of maternal Hoxa-10 expression","volume":"122","author":"Benson","year":"1996","journal-title":"Development"},{"key":"2025021406042414200_DEVELOP_124_23_4781C2","doi-asserted-by":"publisher","first-page":"2293","DOI":"10.1101\/gad.8.19.2293","article-title":"Transcriptional enhancer factor 1 disruption by a retroviral gene trap leads to heart defects and embryonic lethality in mice","volume":"8","author":"Chen","year":"1994","journal-title":"Genes Dev"},{"key":"2025021406042414200_DEVELOP_124_23_4781C3","doi-asserted-by":"publisher","first-page":"473","DOI":"10.1038\/350473a0","article-title":"Regionally restricted developmental defects resulting from targeted disruption of the mouse homeobox gene Hox-1","volume":"350","author":"Chisaka","year":"1991","journal-title":"5. 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