FIGURE

Fig. 6.

ID

ZDB-FIG-200703-6

Publication

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

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Fig. 6.

Ihh signaling within endochondral growth plates is present despite loss of Msmo1 and Lss activity. (A-L) Expression domains of ihha (A,D,G,J), ihhb (B,E,H,K) and ptch1 (C,F,I,L) were analyzed within growth plates of wild type (A-C), kol^nu7 (D-F), Tg(fabp10a:msmo1:pA)^nu100;msmo1^nu81 (G-I) and Tg(fabp10:lss:pA)^nu101;lss^nu60 (J-L) using RNAscope in situ hybridization. (A-C) In wild type, expression of ihha and ihhb is observed in the pre-hypertrophic chondrocyte zone, with ptch1 expression seen to either side of these domains. (D-L) Expression of ihha, ihhb and ptch1 is present within mutant growth plates and similar to domains observed in wild type. Images represent paraffin sections of the pterotic at 2 months of age (∼15 mm SL). Scale bars: 100 μm. Tg*, Tg(fabp10:msmo1:pA)^nu100; Tg**, Tg(fabp10:lss:pA)^nu101. wt n=3, kol^nu7 n=3, kol^nu7/+:msmol^nu81/+ n=3, Tg(fabp10a:msmo1:pA)^nu100;msmo1^nu81 n=3, Tg(fabp10:lss:pA)^nu101;lss^nu60 n=3.

Expression Data

Genes:	ihha ihhb ptch1
Fish:	WT msmo1^nu7/nu7 lss^nu60/nu60; nu101Tg msmo1^nu81/nu81; nu100Tg
Anatomical Terms:	pterotic chondrocyte pterotic hypertrophic chondrocyte
Stage:	Days 45-89

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	msmo1^nu7/nu7 lss^nu60/nu60; nu101Tg msmo1^nu81/nu81; nu100Tg
Observed In:	pterotic smoothened signaling pathway
Stage:	Days 45-89

Phenotype Detail

Acknowledgments

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