ZFIN ID: ZDB-FIG-161228-1
Giunta et al., 2016 - Altered RNA metabolism due to a homozygous RBM7 mutation in a patient with spinal motor neuropathy. Human molecular genetics   25(14):2985-2996 Full text @ Hum. Mol. Genet.
ADDITIONAL FIGURES
PHENOTYPE:
Fish:
Knockdown Reagents:
Observed In:
Stage: Long-pec

Fig. 4

Morphological defects caused by injection of rbm7 MO (A). Mild phenotype fish show slight developmental delay, and shorter body length. Rostrally the head is smaller than control fish while caudally the head looks slightly swollen. Body shape in the moderate phenotype looks compromised and head oedema increased, sporadically we could observe heart oedema as well. Body shape of the severe fish is completely disrupted, swelling of the head is more severe, and we detected smaller forehead and eyes compared with control fish (Scale bar = 200 μm). On the bottom left: RT-PCR of zebrafish cDNA shows efficacy of morpholino against In1-Ex2 boundary. Position of morpholinos is explained in the graph on the right. Below: graphs showing mortality and phenotype rates at 24 hpf. (B) Hindbrain defects in morphant fish. Cranial motor neurons nuclei are well defined in 5 nuclei in wild type fish: Oculomotor neurons (III), Trochlear neurons (IV), Trigeminal neurons (V), Facial neurons (VII), Vagal neurons (X). rbm7-MO seems to have a milder effect on cranial motor-neurons development and to affect predominantly vagal nucleus (nX) which result shorter than controls. Hindbrain structures in exosc8-MO are more generally affected the moderate phenotype fish. Only nX is partially preserved. Exosc3-MO affects predominantly nVII (facial neurons) (Scale bar = 200 μm).

Gene Expression Details No data available
Antibody Labeling Details No data available
Phenotype Details
Fish Conditions Stage Phenotype
rw0Tg + MO1-exosc3 standard conditions Long-pec cranial nerve VII morphology, abnormal
Long-pec hindbrain morphogenesis disrupted, abnormal
Long-pec hindbrain morphology, abnormal
rw0Tg + MO1-rbm7 standard conditions Long-pec hindbrain morphogenesis disrupted, abnormal
Long-pec hindbrain morphology, abnormal
Long-pec motor nucleus of vagal nerve shortened, abnormal
rw0Tg + MO2-exosc8 standard conditions Long-pec hindbrain morphogenesis disrupted, abnormal
Long-pec hindbrain morphology, abnormal
slc24a5unspecified/unspecified + MO1-rbm7 standard conditions Long-pec brain edematous, abnormal
Long-pec embryo development delayed, abnormal
Long-pec eye decreased size, abnormal
Long-pec head swollen, abnormal
Long-pec heart edematous, abnormal
Long-pec whole organism dead, abnormal
Long-pec whole organism decreased length, abnormal
Long-pec whole organism decreased life span, abnormal
Long-pec whole organism deformed, abnormal
Acknowledgments:
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 @ Hum. Mol. Genet.