FNIP1

Cilia effects upon perturbation of FNIP1

Ciliogenesis screen results (2 screens)

• Wheway et al. 2015 (siRNA) [siRNA]: No effect PMID:26167766
• Breslow et al. 2018 (CRISPR) [CRISPR]: No Significant Effect PMID:29459680

Phenotypes

Mouse phenotype:

decreased lung elastance; decreased red blood cell distribution width; prolonged QT interval; small thymus; increased fasting circulating glucose level; decreased locomotor activity; increased lung compliance; decreased grip strength; dilated heart left ventricle; decreased lymphocyte cell number; increased neutrophil cell number; abnormal thymus morphology; increased thigmotaxis; small spleen; enlarged thymus; increased circulating iron level; decreased body weight; decreased effector memory T-helper cell number; decreased lung tissue damping; decreased CD4-positive NK T cell number; increased CD8-positive; alpha-beta T cell number; abnormal inspiratory capacity; decreased respiratory quotient; decreased heart rate; prolonged ST segment; abnormal spleen morphology; increased cardiac stroke volume; abnormal cecum morphology; increased T-helper cell number; increased anxiety-related response; abnormal skeletal muscle morphology; increased freezing behavior; prolonged RR interval; increased T cell number; improved glucose tolerance; decreased airway resistance; decreased lean body mass; abnormal mammary gland morphology; increased heart left ventricle size; increased cardiac muscle contractility; increased CD4-positive; decreased memory-marker CD4-positive NK T cell number; decreased startle reflex; enlarged cecum; decreased spleen weight; decreased effector memory CD8-positive; increased effector memory CD8-positive; decreased exploration in new environment

Mouse ciliopathy phenotype:

enlarged heart; thick ventricular wall; abnormal heart morphology; dilated aorta; increased liver weight; abnormal pancreas morphology; prolonged QRS complex duration; abnormal spine curvature; increased heart weight

Subcellular localization

cilia associated gene

Functional category

• Motile cilium & axoneme; Cilia???cytoskeleton/adhesion links

Function

Depletion of FNIP1, also produced similar effect on mTORC1 activity as did by depletion of FLCN (Fig. 3E), indicating that FNIP1 is involved in the ciliary function of FLCN(PMID: 27072130).