Fig. 1: 15Q11.2 deletion alters network structure.

A, B Representative images of TUJ1⁺ (green, left merged panel) neurons stained for CAMKII (blue) and GAD67 (red) in control and deletion neurons to identify glutamatergic and GABAergic neurons, respectively, demonstrate deficits in network organization. Images taken on day in culture (DIC) 80 A and DIC 146 B. Red arrows indicate GAD67⁺ neurons. Scale bar 50 µm. C Quantification of the percent of TUJ1⁺ neurons expressing CAMKIIα at DIC 80 and 146 presented by donor line with each column representing a donor and each symbol representing an independent culture of that line. D Quantification of the percent of TUJ1⁺ neurons expressing GAD67 at DIC 80 and 146 presented by donor line as in C. Nested one-way ANOVA P = 0.01 with Sidak’s multiple comparison’s test. E Quantification of the percent of TUJ1⁺ neurons expressing GAD67 as well as GAD67⁺ CAMKIIα⁺ and GAD67⁺ CAMKIIα^- neurons. Each symbol represents the mean of a minimum of three cultures from an individual donor line. Bars are the mean ± standard error of the mean (SEM) of four control and three 15q11.2 deletion lines. Two-way ANOVA with Tukey’s multiple comparison correction. F Reverse transcription quantitative PCR (qRT-PCR) data from DIC 141 cultures using human specific primers for interneuron transcripts and the pan-neuronal NKCC1 and KCC2 transcripts. All transcripts were normalized to the expression of the housekeeping gene, human RPS18 (Ribosomal protein S18), and to the mean of the control conditions. Each symbol represents 4 technical replicates for an individual donor’s line. Each bar represents the mean of 4 control and 3 deletion donor lines. Two-way ANOVA with Tukey’s multiple comparison test. * = P < 0.05, ** = P < 0.01, *** = P < 0.001 and **** = P < 0.0001.