TABLE 7.

Zebrafish models of FUS pathology.

Study	Method	Axonopathy	MN loss	NMJ abnormalities	Motor deficits
Armstrong and Drapeau, 2013	Antisense MO knockdown (60% expression reduction)	Not reported	Not reported – But increased MN excitability	Yes – Abnormal structure and deficits in pre- and postsynaptic NMJ transmission	Yes – Reduced swim duration and increased fatigue
Armstrong and Drapeau, 2013	FUS R521H mutation	Not reported	Not reported – But increased MN excitability	Yes – Abnormal structure and deficits in pre- and postsynaptic NMJ transmission	Yes – Reduced swim duration and increased fatigue
Armstrong et al., 2016	CRISPR/Cas9-mediated knockout	Not reported	Not reported	Not reported	Not reported
Kabashi et al., 2011	AMO knockdown	Yes – Reduced primary and unbranched axonal length	Not reported	Not reported	Yes – Deficient TEER (57% of injected)
Kabashi et al., 2011	FUS R521C, R521H, and S57Δ mutations	R521H: Yes – Reduced axonal length. R521C: No S57Δ: No	Not reported	Not reported	R521H: Yes – Deficient TEER (57% of injected). R521C: No S57Δ: No
Lebedeva et al., 2016	CRISPR/Cas9-mediated knockout	No	Not reported	Not reported	No