ZFIN ID: ZDB-FIG-170706-12
Wager et al., 2016 - Neurodegeneration and Epilepsy in a Zebrafish Model of CLN3 Disease (Batten Disease). PLoS One   11:e0157365 Full text @ PLoS One
Knockdown Reagent:
Observed In:
Stage Range: Protruding-mouth to Day 4

Fig. 6

Neurons and glia are disrupted in cln3 ATG MO morphants.

(A-A'', B-B'') Immunohistochemical staining for axons (acetylated α-tubulin) at 4 dpf. (A-A'') normal development of axons in WT larvae. (B-B'') 1.6 ng cln3 ATG MO morphants have a complete absence of axonal organisation throughout the brain, with axonal accumulation (B, dashed arrows), loss of the optic tectum and a narrowing of the optic nerve (B'', dashed arrow). (A-A'', B-B''), anterior to the left; A, B, lateral view, dorsal up; A', B', dorsal view; A'', B'' ventral view. (C-C', D-D') Immunohistochemistry using antibodies to glia (glial fibrillary acidic protein, GFAP) at 4 dpf. (C-C'') Normal staining in WT larvae. (D-D') Ectopic GFAP is observed in the notochord in 1.6 ng cln3 ATG MO morphants (dashed arrow). Lateral view. Anterior to the left. Dorsal up. (E-E', F-F', G-G') Transgenic zebrafish expressing GFP under the control of the HuC promoter in neurons were injected with 1.6 ng cln3 ATG MO and observed at 3 dpf. (E-E') In WT zebrafish, the normal structure of the developing brain and retina can be observed. (F-F') In morphants, there appear to be fewer neurons and the normal brain structure is lost. Many GFP-positive cells were enlarged and found nearer the surface of the brain (F', dashed arrows). (G-G') When the morphology of these enlarged cells was examined further, they lacked typical neuronal morphology. Lateral view. Anterior to the left. Dorsal up. Abbreviations: cb, cerebellum, fb, forebrain; hb, hindbrain; ot, optic tectum; on, optic nerve. A-G'' (all images) Z projection. Scale bars: 100 μm. n = 4 per group.

Gene Expression Details No data available
Antibody Labeling Details No data available
Phenotype Details
Fish Conditions Stage Phenotype
TL + MO1-cln3 standard conditions Day 4 axon extension disrupted, abnormal
Day 4 brain astrocyte increased amount, abnormal
Day 4 brain axon aggregated, abnormal
Day 4 brain axon disorganized, abnormal
Day 4 cranial nerve II decreased width, abnormal
Day 4 optic tectum axon absent, abnormal
Day 4 spinal cord astrocyte increased amount, abnormal
knu3Tg + MO1-cln3 standard conditions Protruding-mouth brain morphology, abnormal
Protruding-mouth brain neuron decreased amount, abnormal
Protruding-mouth neuron morphology, abnormal
This image is the copyrighted work of the attributed author or publisher, and ZFIN has permission only to display this image to its users. Additional permissions should be obtained from the applicable author or publisher of the image. Full text @ PLoS One