FIGURE

Fig. 4.

ID

ZDB-FIG-200703-4

Publication

Anderson et al., 2020 - Zebrafish models of skeletal dysplasia induced by cholesterol biosynthesis deficiency

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Fig. 4.

Loss of Msmo1 and Lss activity is associated with abnormal endochondral bone formation. (A-T) The hyomandibular (A,E,I,M,Q), ceratohyal (B,F,J,N,R) and hypural complex (C,D,G,H,K,L,O,P,S,T) were compared between wild type (A-D), kol^nu7 (E-H), kol^nu7/+:msmol^nu81/+ (I-L), Tg(fabp10a:msmo1:pA)^nu100;msmo1^nu81 (M-P) and Tg(fabp10:lss:pA)^nu101;lss^nu60 (Q-T). (E-T) The endochondral bones of the mutants are compressed and irregularly shaped with ectopic ossification and fusions throughout the growth plates, marked with arrows. Images represent disarticulated whole-mount adult skeletal preparations at ∼4 months of age. Scale bars: 100μm. Tg*, Tg(fabp10:msmo1:pA)^nu100; Tg**, Tg(fabp10:lss:pA)^nu101. wt n=4, kol^nu7 n=4, kol^nu7/+:msmol^nu81/+ n=4, Tg(fabp10a:msmo1:pA)^nu100;msmo1^nu81 n=4, Tg(fabp10:lss:pA)^nu101;lss^nu60 n=4.

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	msmo1^nu7/nu7 msmo1^nu81/+; msmo1^nu7/+ lss^nu60/nu60; nu101Tg msmo1^nu81/nu81; nu100Tg
Observed In:	ceratohyal bone endochondral bone ossification hyomandibula hypural
Stage:	Adult

Phenotype Detail

Acknowledgments

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