FIGURE

Figure 1

ID

ZDB-FIG-210510-1

Publication

Bragazzi Cunha et al., 2021 - Acitretin mitigates uroporphyrin-induced bone defects in congenital erythropoietic porphyria models

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Figure 1

Zebrafish model of CEP develops bone phenotype resembling human disease. (A) 6dpf zebrafish larvae were injected with uro-I or vehicle and imaged by confocal microscopy at 7dpf. Porphyrin was detected only in the bones of uro-I-injected group. Arrowhead-operculum; box-vertebrae. (B) Larvae were treated as in (A) and injected with calcein prior to imaging. Arrowhead-operculum; arrow-4^th vertebra. (C) Quantification of bone volume in larvae from (B); bone volume was normalized to vehicle-injected larvae set to 100%. Symbols represent individual larvae (14–18/group) from 4–5 independent experiments. (D) Larvae were treated as in (A). At 7dpf bones were harvested and imaged by epifluorescence microscopy pre and post HCl bone demineralization, arrow-notochord. (E) Hydroxyapatite was incubated with calcein/uro-I/copro-I and imaged by epifluorescence microscopy. Scale bars: 200 µm (A-D); 50 µm (E). Three-dimensional image reconstruction (A, B) was performed using Imaris 3D software v7.7 (http://imaris.oxinst.com/). **p < 0.01.

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	WT
Condition:	chemical treatment by injection: uroporphyrin I
Observed In:	bone tissue porphyrin opercular flap vertebra
Stage:	Days 7-13

Phenotype Detail

Acknowledgments

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