biostudies-literatureRieke JMDeutsche ForschungsgemeinschaftMedical Research CouncilWellcome Trust567https://www.ebi.ac.uk/biostudies/studies/S-EPMC7426641biostudies-literatureUnknown8Previous studies in developing <i>Xenopus</i> and zebrafish reported that the phosphate transporter <i>slc20a1a</i> is expressed in pronephric kidneys. The recent identification of <i>SLC20A1</i> as a monoallelic candidate gene for cloacal exstrophy further suggests its involvement in the urinary tract and urorectal development. However, little is known of the functional role of <i>SLC20A1</i> in urinary tract development. Here, we investigated this using morpholino oligonucleotide knockdown of the zebrafish ortholog <i>slc20a1a</i>. This caused kidney cysts and malformations of the cloaca. Moreover, in morphants we demonstrated dysfunctional voiding and hindgut opening defects mimicking imperforate anus in human cloacal exstrophy. Furthermore, we performed immunohistochemistry of an unaffFrontiers in cell and developmental biology&lt;i&gt;SLC20A1&lt;/i&gt; Is Involved in Urinary Tract and Urorectal Development.PMC7426641MR/R006237/11172/37-1RE 1723/1-1MR/L002744/1OD 102/1-3RE 1723/1-3TH 1327/1-1Schmiedeke EBarker GBeaman GMZhang RPromm MMarcelis CLMGrote PSharma AHolmdahl GKeegan CEBraun DFeitz WFJVan Rooij IALMRosch WHBrusco AEbert AKJapp ASZwink NBokenkamp ABeckers GMADi Grazia MLudwig MNordenskjold ALacher MLackgren GHirsch KAnderberg MPleschka MStein RBoemers TMJenetzky EThiele HKluth DKeene DWittler LSchafer FMOdermatt BGosemann JHHilger ACRieke JMGiorgio ELopes FMSchneider SWoolf ASReutter HYilmaz ONewman WGCervellione RMSchweizer UDakal TCfalse&lt;i&gt;SLC20A1&lt;/i&gt; Is Involved in Urinary Tract and Urorectal Development.Previous studies in developing <i>Xenopus</i> and zebrafish reported that the phosphate transporter <i>slc20a1a</i> is expressed in pronephric kidneys. The recent identification of <i>SLC20A1</i> as a monoallelic candidate gene for cloacal exstrophy further suggests its involvement in the urinary tract and urorectal development. However, little is known of the functional role of <i>SLC20A1</i> in urinary tract development. Here, we investigated this using morpholino oligonucleotide knockdown of the zebrafish ortholog <i>slc20a1a</i>. This caused kidney cysts and malformations of the cloaca. Moreover, in morphants we demonstrated dysfunctional voiding and hindgut opening defects mimicking imperforate anus in human cloacal exstrophy. Furthermore, we performed immunohistochemistry of an unaff2020-01-01T00:00:00Z20202026-05-03T20:41:48.495Z2020-08-29T07:23:38ZS-EPMC74266413285077810.3389/fcell.2020.00567