FIGURE

Fig. 5

ID

ZDB-FIG-230831-17

Publication

Jackson et al., 2023 - Clinical, genetic, epidemiologic, evolutionary, and functional delineation of TSPEAR-related autosomal recessive ectodermal dysplasia 14

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Fig. 5

Zebrafish tspear double-knockout model

(A) A schematic figure showing ventral view of larval zebrafish with ventral cartilages (blue) and teeth (red). Alizarin red S staining of pharyngeal teeth shows that a double homozygous mutant (tspeara^−/−;tspearb^−/−) displayed thinner and aberrant mineralized teeth (arrowhead) at 12 days post-fertilization (dpf).

(B) Schematic figure for pharyngeal dentition of adult zebrafish shown in ventral view (top panel) and lateral view (bottom panel). Arrow indicates missing tooth. A, anterior; D, dorsal; MD, mediodorsal; V, ventral.

(C) Quantification of the number of teeth in the ventral, mediodorsal, and dorsal rows. Each dot represents one animal. Control = 30 animals, mutant = 32 animals. Error bars: mean ± SD. Difference was tested using two-tailed unpaired t test with Mann-Whitney test. ∗∗∗p < 0.001, ∗∗∗∗p < 0.0001.

(D) Adult tail fins were amputated from wild-type control (n = 4) and double homozygous mutants (n = 4). Live fins were imaged at 0, 5, and 12 days post-amputation (dpa). Dashed line indicates the position of amputation.

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	tspearb^{omf107/omf107}; tspeara^{omf106/omf106}
Condition:	amputation: caudal fin
Observed In:	caudal fin fin regeneration ceratobranchial 5 tooth ceratobranchial 5 tooth bone mineralization
Stage Range:	Days 7-13 to Adult

Phenotype Detail

Acknowledgments

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