FIGURE

Fig. 3

ID

ZDB-FIG-180913-66

Publication

de Vos et al., 2018 - Functional analysis of a hypomorphic allele shows that MMP14 catalytic activity is the prime determinant of the Winchester syndrome phenotype

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Fig. 3

The mmp14a/b KO zebrafish recapitulate key aspects of the WS phenotype. (A) Gross anatomy photographs of 3-month-old WT and mmp14a/b KO fish of respective average size; lateral view, anterior to the left. The phenotype of mmp14a/b KO fish includes a relatively small, up-tilted head with relatively large, protruding eyes and a short operculum. Limited field of view necessitated stitching of multiple photographs together, causing the vertical line in the images shown in (A). Scale bar equals 2 mm. (B) At 90 dpf, mmp14a/b KO fish have a significantly shorter total body length compared with WT fish (P < 0.0001). A minimum of 21 individuals per genotype was measured. (C) 3D reconstruction of µCT scans of 3-month-old WT and mmp14a/b KO fish; lateral view, anterior to the left. Compared with WT fish, the mmp14a/b KO fish have Weberian-prehemal hyperkyphosis. (D) The mmp14a/b KO fish have a reduced skull bone mineral density (BMD, P < 0.05), giving the appearance of missing skeletal elements in the shown 3D reconstruction (C). BMD was assessed for five individuals per genotype. The individuals imaged in (C) are different from the ones shown in (A).

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	mmp14a^{zf2194/zf2194}; mmp14b^{zf2195/zf2195} (AB)
Observed In:	cranium cranium bone mineralization eye head non-Weberian precaudal vertebra opercular flap vertebral column Weberian vertebra whole organism
Stage:	Adult

Phenotype Detail

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.