Reviewer 2's Review
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The authors present a well-carried out study of activation of cAMP production by forskolin in two FXS model organisms and in human FXS neural cells. The goal of the studies presented in this report was to confirm prior findings of abnormal cAMP production in FXS, related to deficits in FMRP expression, and extend those findings to FXS models and FXS neural cells in culture, none of which were available at the time of the original studies. The results confirm the defect in peripheral and neural tissues in the FXS k/o mouse and neural tissue from all three species, as well as rescue of the defect by reintroduction of dFXR into the Drosophila model. The data is well-presented and the study is well described.
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N.B. These are the general comments made by the reviewer when reviewing this paper in light of which the manuscript was revised. Specific points addressed during revision of the paper are not shown.
