Fig 1: Neuropeptide production and release are impaired in Magel2p?/m+ mice and human PWS iN.(A) Peptide abundance of indicated neuropeptides in 8-week-old Magel2+/+ and Magel2p?/m+ mouse hypothalamuses. Each data point represents 1 animal, plotted as mean ± SD (n = 5 per genotype) and indicated P values as analyzed by unpaired, 2-tailed t test with Bonferroni’s correction. (B–F) Reduced levels of plasma OXT (B), AVP (C), SST (D), GnRH (E), and a-MSH (F) in 14-week-old Magel2p?/m+ mice, measured by ELISA. Each data point represents 1 animal, plotted as mean ± SD (n > 5 per genotype), and analyzed by unpaired, 2-tailed t test. (G) ELISA analysis of oxytocin levels in human iN media, at basal level and after KCl (50 mM, 30 minutes) stimulation. Each data point represents 1 induction experiment, plotted as mean ± SD (n = 3), and analyzed by 1-way ANOVA. (H) Transcript levels of various neuropeptides between 8-week-old Magel2p?/m+ and Magel2+/+ mouse hypothalamuses. Each data point represents 1 animal, plotted as mean ± SD (n = 3 per genotype) and analyzed by unpaired, 2-tailed t test. (I) Transcript levels of various neuropeptides between control and PWS iN at 14 days postinduction. Each data point represents 1 induction experiment (n = 2), plotted as mean ± SD and analyzed by 1-way ANOVA. (J) Reduced levels of plasma CHGB in PWS patients after 12 hours of fasting, measured by ELISA. Each data point is a unique individual, plotted as mean ± SD and analyzed by Mann-Whitney U test. *P < 0.05, ***P < 0.001, and ****P < 0.001.