BMP4

Cilia effects upon perturbation of BMP4

Cilia number / % ciliated:

Decreased cilia number

Ciliogenesis screen results (4 screens)

• Wheway et al. 2015 (siRNA) [siRNA]: Ciliogenesis Defect (z=-2.62) PMID:26167766
• Breslow et al. 2018 (CRISPR) [CRISPR]: No Significant Effect PMID:29459680
• Roosing et al. 2015 (siRNA) [siRNA]: No effect PMID:26595381
• Pusapati et al. 2018 (CRISPR) [CRISPR]: Positive Regulator (Hh signaling, neg_rank=132, lfc=-2.31) PMID:30270045

Phenotypes

Mouse phenotype:

abnormal hindbrain development, hyperactivity, abnormal embryo size, abnormal reti inner nuclear layer morphology, abnormal forebrain development, abnormal optic disk morphology, abnormal allantois morphology, abnormal eye anterior chamber depth, decreased anxiety-related response, increased grip strength, preweaning lethality, complete penetrance, eyelids fail to open, decreased total reti thickness, abnormal reti outer nuclear layer morphology, abnormal embryo development, abnormal iris morphology, embryonic growth retardation, embryonic lethality prior to tooth bud stage

Mouse ciliopathy phenotype:

abnormal embryo turning, abnormal neural tube closure, abnormal heart morphology, abnormal midbrain development, microphthalmia, abnormal reti morphology

Human ciliopathy phenotype:

microphthalmia with brain and digit anomalies

Subcellular localization

cilia associated gene

Functional category

• Ciliary assembly/disassembly
• Actin & cytoskeleton regulation
• T cell biology
• Cell migration & adhesion
• Signaling (Hedgehog, GPCRs, ion channels)
• Cardiac & muscle development
• ECM & connective tissue
• Transcription regulation

Function

Supplementation of BMP2 or BMP4 in culture media significantly reduced the number of cells with multiple cilia among the total cells, while most of the cells expressed FoxJ1, a master regulator of ciliogenesis( 33087680).

Model organism evidence