Table 1.

Selected studies investigating neuropsychiatric disorders using hiPSCs from patients.

Disease	Type of cells	Genetic variants	Phenotypes	Reference
Rett syndrome	Neurons	MECP2	Fewer synapses, reduced spine density, smaller soma size, altered calcium signaling, electrophysiological defects	(Marchetto et al., 2010)
Rett syndrome/ASD	Neurons	CDKL5	Aberrant dendritic spines	(Ricciardi et al., 2012)
Rett syndrome	NPCs	MECP2	Migration defects in layered 3D culture; reduced neurite outgrowth and fewer synapses	(Zhang et al., 2016)
Non-syndromic ASD	Neurons	TRPC6	Altered neuronal development, morphology and function	(Griesi-Oliveira et al., 2015)
ASD	Organoids	Idiopathic	Accelerated cell cycle, overproduction of GABAergic inhibitory neurons	(Mariani et al., 2015)
ASD	NPCs, Neurons	Idiopathic	Increased cell proliferation of NPCs, abnormal neurogenesis, reduced synaptogenesis, defects in neuronal networks	(Marchetto et al., 2017)
ASD	Neurons	16p11.2 deletion/ 16p11.2 duplication	Increased soma size and dendrite length in 16pdel neurons; reduced neuronal size and dendrite length in 16pdup neurons	(Deshpande et al., 2017)
ASD	Cortical neurons	Idiopathic	Morphological growth acceleration of early neuron development	(Schafer et al., 2019)
Timothy syndrome	Cortical neurons	CACNA1C	Abnormal expression of tyrosine hydroxylase and increased production of norepinephrine and dopamine	(Pasca et al., 2011)
Timothy syndrome	Subdomain specific forebrain spheroids	CACNA1C	Abnormal migratory saltations	(Birey et al., 2017)
Fragile X syndrome	Neurons	FMR1	Abnormal homeostatic synaptic plasticity	(Zhang et al., 2018)
Bipolar disorder	NPCs	Idiopathic	Increased CXCR4 expression; phenotypic differences in neurogenesis	(Madison et al., 2015)
Bipolar disorder	DG neurons	Idiopathic	Mitochondrial abnormalities, hyperactive action-potential firing	(Mertens et al., 2015)
Bipolar disorder	Neurons	Idiopathic	Increased expression of miR-34a	(Bavamian et al., 2015)
Bipolar disorder	DG neurons	Idiopathic	Hyperactive action-potential firing, fast after-hyperpolarization	(Stern et al., 2018)
SZ	Neurons	Idiopathic	Diminished neuronal connectivity, decreased neurite number	(Brennand et al., 2011)
SZ	DG neurons	Idiopathic	Deficits in the generation of DG neurons, reduced neuronal activity, reduced spontaneous neurotransmitter release	(Yu et al., 2014)
SZ	NPCs	15q11.2 microdeletion	Deficits in adherens junctions and apical polarity	(Yoon et al., 2014)
SZ	Neurons	Idiopathic	Elevated levels of dopamine, norepinephrine, and epinephrine secretion	(Hook et al., 2014)
SZ	Neurons	22q11 deletion	Increase L1 copy number	(Bundo et al., 2014)
SZ	NPCs	Idiopathic	Higher variability in the levels of HSF1 activation induced by environmental challenges	(Hashimoto-Torii et al., 2014)
SZ	Forebrain neurons	DISC1	Synaptic vesicle release deficits	(Wen et al., 2014)
SZ	NPCs	Idiopathic	Abnormal gene expression and protein levels related to cytoskeletal remodeling and oxidative stress	(Brennand et al., 2015)
SZ	Neurons	22q11.2 microdeletion	45 differentially expressed miRNAs	(Zhao et al., 2015a)
SZ	NPCs	DISC1	Increased miR-219 expression	(Murai et al., 2016)
SZ	Glial progenitors/humanized glial chimeric mice	Idiopathic	Premature migration into the cortex; delayed astrocytic differentiation and abnormal astrocytic morphologies	(Windrem et al., 2017)
SZ	CA3 neurons, DG neurons	Idiopathic	Reduced activity in DG-CA3 co-culture, deficits in spontaneous and evoked activity in CA3 neurons	(Sarkar et al., 2018)
SZ	Cortical interneurons	Idiopathic	Defects in synaptic density and arborization	(Shao et al., 2019)
SZ	Microglia and neurons	Idiopathic	Increased synapse elimination in neural cultures and isolated synaptosomes	(Sellgren et al., 2019)
AxD	Astrocytes and OPCs	GFAP	GFAP aggregates and Rosenthal fibers; AxD astrocytes inhibit OPC proliferation and myelination	(Li et al., 2018)
AxD	Astrocytes	GFAP	GFAP aggregates; impaired extracellular ATP release; enlarged and heterogeneous morphology coupled with perinuclear localization of endoplasmic reticulum and lysosomes	(Jones et al., 2018)
AxD	Astrocytes	GFAP	Increased expression of A-type lamin and nuclear yes-associated protein; increased numbers of stress fibers in the soma of AxD astrocytes	(Wang et al., 2018)
Phelan-McDermid syndrome	Neurons	22q13 deletion	Major defects in excitatory but not inhibitory synaptic transmission	(Shcheglovitov et al., 2013)
Major depressive disorder	Forebrain neurons	Idiopathic	Serotonin-induced hyperactivity downstream of upregulated excitatory serotonergic receptors in selective serotonin reuptake inhibitors-nonremitters	(Vadodaria et al., 2019)