A) Schematic for two methods to derive microglia from PMACS. i) Day 10 microglial progenitors are co-cultured with postmitotic hPSC-derived neurons for 1 week in the presence of IL-34 and M-CSF. ii) Day 10 microglial progenitors are matured into primitive macrophages with IL-34 and M-CSF, then co-cultured with hPSC-derived neurons for 4 days. B)i. IF shows ramified IBA1+PU.1+ cells after day 5 of direct co-culture of day 10 progenitors with postmitotic hPSC-derived cortical neurons. Scale bar = 50 μM. ii. IF shows IBA1+ cells distributed evenly throughout the neuronal culture by day 10 of co-culture. Scale bar = 200uM. C) FACS analysis shows that over 30% of cells in co-culture at day 5 express CX3CR1+. D) FACS analysis shows that GFP+ day 10 progenitors co-cultured with hPSC-derived cortical neurons for 6 days are ~84% CD45+ indicating commitment to the microglial lineage. Of these over 80% are CX3CR1+, indicating maturation into early microglia. The ~15% GFP+ population that is CD45- is ~50% CD41+CD235A+ (primitive EMPs), and ~50% uncommitted. E) FACS analysis shows that maturing Day 10 progenitors in IL-34 and M-CSF without neurons yields a progressively pure population of primitive macrophages expressing CD11B (~99%) and CX3CR1 (>85%) by 11 days in culture. PBMCs matured in parallel express CD11B (100%) but not CX3CR1+ at day 11. F) IF shows ramified IBA1+ microglial-like cells after culturing primitive macrophages with hPSC-derived cortical neurons for 4 days. Scale bar = 50 μM. G) FACS analysis shows that co-cultured microglial cells maintain expression of CX3CR1 and have a lower expression of CD45 than PBMC-matured macrophages co-cultured with cortical neurons, which largely do not express CX3CR1 and have higher CD45 expression. H) qPCR of a panel of microglial-specific genes shows that hPSC-microglia generated from either method express these genes at similar levels to human primary microglial cDNA (Celprogen, commercially available), whereas PBMC-derived macrophages do not. Fold change is over day 10 progenitors for CX3CR1, TMEM119, C1QA, GPR34, and over hESCs for P2RY12. n=2 technical replicates of a representative qPCR. I) Bulk RNA-sequencing of microglia derived from 2 different methods (Mac-cocl = method ii, matured alone then co-cultured, Direct-cocl = method i, direct co-culture) show similarity to each other and to acutely isolated adult primary microglia from postmortem samples. hPSC-derived neurons sorted from co-culture grouped separately. n=3 samples per group. J) i) Confocal imaging shows microglia co-cultured with d70 hPSC-derived cortical neurons for 30 days contain inclusions of postsynaptic proteins (PSD95). Scale bar = 50 μM. ii) Quantification of inclusions show that inclusions containing general neuronal matter (tagged with RFP) are greater in volume than inclusions containing postsynaptic protein (PSD95). n=4 fields. Error bars = SD, center = mean.