In this study, we determine that the tubby family protein TULP3 functions as a general adapter for ciliary trafficking of structurally diverse integral membrane cargo, including multiple reported and novel rhodopsin family G protein-coupled receptors (GPCRs) and the polycystic kidney disease-causing polycystin 1/2 complex.
Mice homozygous for a hypomorphic missense mutation within the conserved Tubby domain of Tulp3 develop cysts at late embryonic stages, leading to severe postnatal loss of kidney function.
In addition, higher TULP3 gene expression was associated to lymphatic and vascular invasion in colon adenocarcinoma (COAD) and rectum adenocarcinoma (READ), respectively.
In addition, higher TULP3 gene expression was associated to lymphatic and vascular invasion in colon adenocarcinoma (COAD) and rectum adenocarcinoma (READ), respectively.
However, in an adult model of ADPKD utilizing inducible conditional Pkd1 deletion, concomitant removal of Tulp3 surprisingly ameliorates cystic disease.
In addition, higher TULP3 gene expression was associated to lymphatic and vascular invasion in colon adenocarcinoma (COAD) and rectum adenocarcinoma (READ), respectively.
In addition, higher TULP3 gene expression was associated to lymphatic and vascular invasion in colon adenocarcinoma (COAD) and rectum adenocarcinoma (READ), respectively.
We have identified a new mouse mutant that is a strongly hypomorphic allele of Tulp3 and which exhibits expansion of ventral markers in the caudal spinal cord, as well as neural tube defects and preaxial polydactyly, consistent with increased Shh signalling.
Mouse hitchhiker mutants have spina bifida, dorso-ventral patterning defects and polydactyly: identification of Tulp3 as a novel negative regulator of the Sonic hedgehog pathway.
We have identified a new mouse mutant that is a strongly hypomorphic allele of Tulp3 and which exhibits expansion of ventral markers in the caudal spinal cord, as well as neural tube defects and preaxial polydactyly, consistent with increased Shh signalling.