ArticleDevelopmental biology2011
Six1 and Eya1 are critical regulators of peri-cloacal mesenchymal progenitors during genitourinary tract development.
Article in Developmental biology, 2011. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 22 papers, 1 of them a synthesis that pooled it.
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Who cites it
22 citing papers in PubMed, 1 synthesis or guideline pooled it, 41 citations in OpenAlex.
- Unraveling the Androgen Receptor's Role in Hypospadias: A Systematic Review and Meta-Analysis.International journal of molecular sciences · 2026Pooled it
- The cloacal outgrowth orchestrates co-development of the bladder and umbilical arteries.Development (Cambridge, England) · 2026Article
- An extra-genital cell population contributes to urethra closure during mouse penis development.bioRxiv : the preprint server for biology · 2023Article
- Distal vaginal atresia: a report of a rare type found a late-term fetus and its histological comparison with the normal pelvis.Anatomy & cell biology · 2022Article
- One Tool for Many Jobs: Divergent and Conserved Actions of Androgen Signaling in Male Internal Reproductive Tract and External Genitalia.Frontiers in endocrinology · 2022Review
- The SIX Family of Transcription Factors: Common Themes Integrating Developmental and Cancer Biology.Frontiers in cell and developmental biology · 2021Review
- Meeting report on the NIDDK/AUA Workshop on Congenital Anomalies of External Genitalia: challenges and opportunities for translational research.Journal of pediatric urology · 2020Review
- Renal developmental genes are differentially regulated after unilateral ureteral obstruction in neonatal and adult mice.Scientific reports · 2020Article
- Temporally Distinct Six2-Positive Second Heart Field Progenitors Regulate Mammalian Heart Development and Disease.Cell reports · 2017Article
- Article
- Clarification of mammalian cloacal morphogenesis using high-resolution episcopic microscopy.Developmental biology · 2016Article
- An illustrated anatomical ontology of the developing mouse lower urogenital tract.Development (Cambridge, England) · 2015Article
- A mouse model of urofacial syndrome with dysfunctional urination.Human molecular genetics · 2015Article
- The great divide: septation and malformation of the cloaca, and its implications for surgeons.Pediatric surgery international · 2014Review
- Genome-wide association analyses identify variants in developmental genes associated with hypospadias.Nature genetics · 2014Article
- Defining the molecular pathologies in cloaca malformation: similarities between mouse and human.Disease models & mechanisms · 2014Article
- Dkk1 in the peri-cloaca mesenchyme regulates formation of anorectal and genitourinary tracts.Developmental biology · 2014Article
- Article
- Asymmetric requirement of surface epithelial β-catenin during the upper and lower jaw development.Developmental dynamics : an official publication of the American Association of Anatomists · 2012Article
- Article
Corrections and comments
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Authors and funding
7 authors at 1 institution in 1 country.
Funding
Abstract
The evolutionarily conserved Six1-Eya1 transcription complex is central to mammalian organogenesis, and deletion of these genes in mice results in developmental anomalies of multiple organs that recapitulate human branchio-oto-renal (BOR) and DiGeorge syndromes. Here, we report that both Six1 and Eya1 are strongly expressed in the peri-cloacal mesenchyme (PCM) surrounding the cloaca, the terminal end of hindgut dilation. Six1 and Eya1 are absent from the intra-cloacal mesenchyme (ICM), a cell mass that divides the cloaca into dorsal hindgut and ventral urogenital sinus. Deletion of either or both Six1 and Eya1 genes results in a spectrum of genitourinary tract defects including persistent cloaca - hypoplastic perineum tissue between external urogenital and anorectal tracts; hypospadias - ectopic ventral positioning of the urethral orifice; and hypoplastic genitalia. Analyses of critical signaling molecules indicate normal expression of Shh in the cloaca and cloaca-derived endodermal epithelia. Using a Cre/loxP genetic fate mapping strategy, we demonstrate that Six1-positive PCM progenitors give rise to the most caudal structures of the body plan including the urogenital and anorectal complex, and the perineum region. Thus, Six1 and Eya1 are key regulators of both upper and lower urinary tract morphogenesis. Results from this study uncover essential roles of the PCM progenitors during genitourinary tract formation.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.