Table 1.

Genes essential for DFC/KV organogenesis

Loss-of-function	Gene	Protein	DFCs	KV	KV ciliogenesis	References
Mutation	aei/deltaD	Notch ligand	n.d.	Normal	Shortened	[30]
Mutation	cas/sox32	TF induced by Nodal	No DFCs	No KV	No cilia	[14]
Mutation	oep	Nodal co-receptor	No DFCs	No KV	No cilia	[14]
DFC-KD; KD	Ttrap	Nodal antagonist	Spread	n.d.	n.d	[13]
KD	frizzled-2	nc-Wnt receptor	n.d.	Normal	Shortened/reduced	[36]
KD	rock2b	nc-Wnt mediator	n.d.	Small lumen	Abnormal cilium arrangement	[47]
KD	prickle1a	nc-Wnt mediator	Failed compaction	Fragmented lumens	Shortened	[38]
Mutation	MZvangl2	nc-Wnt mediator	n.d.	Expanded	Abnormal cilium orientation	[7]
Mutation	MZkny	nc-Wnt mediator	n.d.	Expanded	Abnormal cilium orientation	[7]
KD	duboraya	nc-Wnt mediator	n.d.	Normal	Shortened/reduced	[36]
KD	foxj1a	TF induced by Hedgehog	n.d.	Small	Shortened/reduced	[45]
DFC-KD	no tail	TF induced by FGF	n.d.	Disorganized	Shortened/reduced	[5]
DFC-KD; KD	tbx16	TF induced by FGF	Broken-up clusters	Small/disrupted	Reduced	[4, 31]
DFC-KD; KD	fgfr1a	FGF receptor	Normal	Normal	Shortened	[35]
DFC-KD; KD	canopy1	FGF positive regulator	Broken-up clusters	Small/disrupted	Shortened/reduced	[31]
KD	ire1	FGF mediator	Broken-up clusters	Small/disrupted	Shortened/reduced	[20]
KD	fibp1	FGF mediator	Broken-up clusters	Small/disrupted	Shortened/reduced	[20]
Mutation	ace/fgf8	FGF ligand	Broken-up clusters	Small/disrupted	Reduced	[3, 31, 35]
DFC-KD	cadherin1	Adhesion molecule induced by FGF	Broken-up clusters	Small/disrupted	Shortened/reduced	[13, 31, 38]
KD	CaMK-II	Ca2+ signal mediator	n.d.	Small	Shortened/reduced	[16]
DFC-KD; KD	lrd	IFT component	Normal	Normal	Non-motile	[14, 23]
KD	ift57	IFT component	n.d.	n.d.	No/shortened cilia	[25]
KD	ift88	IFT component	n.d.	n.d.	No/shortened cilia	[25]
DFC-KD; KD	connexin43.4	Gap junction component	n.d.	Small	Shortened	[18]
KD	miR-92	Fine-tuner of Gata5	n.d.	Small/disrupted	Shortened/reduced	[28]
DFC-KD; KD	integrin a5	Adhesion molecule to ECM	Broken-up clusters	Small	Shortened/reduced	[1]
KD	nde1	Centrosomal protein	n.d.	Small	Elongated	[24]

DFC-KD DFC-specific knockdown, KD knockdown, TF transcription factor, n.d. not determined