Fig. 1. Rescue of the Inv/Inv phenotype by expression of an Inv::GFP transgene. (A) Schematic representation of the structure of the Inv::GFP fusion protein. IQI and IQII represent two putative calmodulin binding motifs present in Inv. (B) Expression of the Inv::GFP fusion protein in a mouse embryo at embryonic day (E) 12.5. GFP fluorescence was detected in the transgenic embryo shown on the right but not in the non-transgenic littermate shown on the left. (C-G) Comparison of Inv/Inv mice with or without the Inv::GFP transgene at postnatal day 2. (C) Fluorescence was detected in the forelimb of an Inv/Inv, Inv::GFP mouse (right) but not in that of an Inv/Inv mouse without the transgene (left). (D) The stomach (arrowhead) was located on the right side of an Inv/Inv mouse (left embryo) but on the left side of an Inv/Inv, Inv::GFP mouse (right embryo). (E) Lung lobation pattern of an Inv/Inv mouse (left) and an Inv/Inv, Inv::GFP mouse (right). L, left; R, right. (F,G) The kidneys of an Inv/Inv mouse (F) exhibited multiple cysts (arrowheads) that were not present in the Inv/Inv, Inv::GFP mouse (G). Kidney sections were stained with Hematoxylin and Eosin.

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