TABLE 6.

Zebrafish models of TARDBP (TDP-43) pathology.

Study	Method	Axonopathy	MN loss	NMJ abnormalities	Motor deficits	Other phenotypes
Bose et al., 2019	CRISPR/Cas9-mediated knockout	Not reported	Not reported	Yes – Abnormal structure and deficits in pre- and postsynaptic NMJ transmission	Yes – Reduced swim duration, distance, and maximum velocity	Morphological deformities. Increased mortality.
Hewamadduma et al., 2013	TILLING-mediated TARDBP fh301 (Y220) missense mutation	No (not in tardbpfh301/fh301 mutants) – But severe axonal defects in double (tardbp + tardbpl) knockouts	Not reported	Not reported	Yes – In double (tardbp + tardbpl) knockout	Morphological abnormalities and increased mortality in double (tardbp + tardbpl) knockouts
Kabashi et al., 2010	AMO knockdown	Yes – Reduced axon length and aberrant branching	No	Not reported	Yes – Impaired tail coiling ability and loss of TEER
Kabashi et al., 2010	TARDBP A315T, G348C, and A382T mutations	A315T: Yes – Reduced axon length and increased branching. G348C: Yes – Reduced axon length and increased branching. A382T: Yes – Reduced axon length	A315T: Yes – 48%. G348C: Yes – 44%. A382T: Yes – 31%.	Not reported	Yes – Impaired tail coiling ability and delayed response to TEER
Laird et al., 2010	TARDBP A315T mutation	Yes – Reduced axon length and increased aberrant branching	Not reported	Not reported	Not reported
Lissouba et al., 2018	TARDBP G348C mutation	Yes – Increased aberrant branching of primary axon and absence of secondary branching	Not reported	Not reported	Yes – Deficient TEER; reduced swim distance, duration, and maximum velocity
Schmid et al., 2013	TARDBP double (tardbp–/–; tardbpl–/–) mutation	Yes – Reduced axon length	Not reported	Not reported	Not reported	Impaired blood circulation. Muscle atrophy. Increased mortality.