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ZFIN ID: ZDB-FIG-140909-32
Apschner et al., 2014 - Pathological mineralization in a zebrafish enpp1 mutant exhibits features of Generalized Arterial Calcification of Infancy (GACI) and Pseudoxanthoma Elasticum (PXE). Disease models & mechanisms   7(7):811-22 Full text @ Dis. Model. Mech.
ADDITIONAL FIGURES
EXPRESSION / LABELING:
Gene:
Fish:
Anatomical Terms:
Stage Range: Day 6 to Days 7-13
PHENOTYPE:
Fish:
Observed In:
Stage Range: Day 4 to Days 7-13

Fig. 6

Cells expressing osteoclastic markers appear at ectopic mineralization sites in dgf mutants. Staining of Trap in a wild-type sibling (A) and dgf embryo (B). Trap staining (red) was visible in a dgf embryo at the region of the heart and yolk sac (B, arrowhead). Ventral view of sibling (C) and mutant (D) embryo with ectopic mineralization in the heart and yolk sac region. (C) No cathepsinK-positive cells were visible in siblings at this timepoint; (D) mutants showed colocalization of ectopic soft tissue mineralization and cathepsinK-positive cells, but no cathepsinK-positive cells were aligned to skeletal elements, such as the cleithrum (cl). (E,F) Ventral view of the yolk sac area of embryos that had been stained for Trap and with an antibody against GFP (α GFP). (F) Trap staining appeared in association with cathepsinK-positive cells. White arrows indicate loci of osteoclasts with high Trap activity. (G) Higher-magnification image of cathepsinK and Trap colocalization in a dgf mutant. Scale bar: 20μm. The staining of Trap in E–G is pseudo-colored for improved visibility. In the axial skeleton, cathepsinK-positive cells were not visible in siblings (H); however, in dgf mutants (I), cathepsinK-positive cells appeared from 9 dpf onwards and colocalized with mineralized vertebral bodies. White arrows indicate osteoclasts aligning with vertebral elements. (J) Ventral view of accumulating cathepsinK-positive cells in the skin of the heart region. These cells also express the macrophage marker mpeg1.

Gene Expression Details
Gene Antibody Fish Conditions Stage Qualifier Anatomy Assay
Citrine enpp1hu4581/hu4581; zf336Tg standard conditions Day 6 Not Detected cleithrum IHC
Day 6 heart IHC
Day 6 integument IHC
Days 7-13 integument osteoclast IHC
Days 7-13 vertebra osteoclast IHC
zf336Tg standard conditions Day 6 Not Detected cleithrum IHC
Days 7-13 Not Detected integument IHC
Days 7-13 Not Detected integument osteoclast IHC
Antibody Labeling Details No data available
Phenotype Details
Fish Conditions Stage Phenotype
enpp1hu4581/hu4581 standard conditions Day 4 ball integument calcified, abnormal
Day 4 bone mineralization process quality, abnormal
Day 4 heart calcified, abnormal
enpp1hu4581/hu4581; zf336Tg standard conditions Day 6 ball integument calcified, abnormal
Day 6 bone mineralization process quality, abnormal
Day 6 heart calcified, abnormal
Days 7-13 ball integument calcified, abnormal
Days 7-13 bone mineralization process quality, abnormal
Days 7-13 integument osteoclast mislocalised, abnormal
Days 7-13 vertebra bone mineralization increased occurrence, abnormal
Days 7-13 vertebra bone resorption increased occurrence, abnormal
Days 7-13 vertebra osteoclast mislocalised, abnormal
Acknowledgments:
ZFIN wishes to thank the journal Disease models & mechanisms for permission to reproduce figures from this article. Please note that this material may be protected by copyright. Full text @ Dis. Model. Mech.