Abstract
Background
   Sexual dimorphism is highly prominent in mammals with many
   physiological and behavioral differences between male and female form
   of the species. Accordingly, the fundamental social and cultural
   stratification factors for humans is sex. The sex differences are
   thought to emerge from a combination of genetic and environmental
   factors. It distinguishes individuals most prominently on the
   reproductive traits, but also affects many of the other related traits
   and manifest in different disease susceptibilities and treatment
   responses across sexes. Sex differences in brain have raised a lot of
   controversy due to small and sometimes contradictory sex-specific
   effects. Many studies have been published to identify sex-biased genes
   in one or several brain regions, but the assessment of the robustness
   of these studies is missing. We therefore collected huge amount of
   publicly available transcriptomic data to first estimate whether
   consistent sex differences exist and further explore their likely
   origin and functional significance.
Results and conclusion
   In order to systematically characterise sex-specific differences across
   human brain regions, we collected transcription profiles for more than
   16,000 samples from 46 datasets across 11 brain regions. By systematic
   integration of the data from multiple studies, we identified robust
   transcription level differences in human brain across to identify
   male-biased and female-biased genes in each brain region. Firstly, both
   male and female-biased genes were highly conserved across primates and
   showed a high overlap with sex-biased genes in other species.
   Female-biased genes were enriched for neuron-associated processes while
   male-biased genes were enriched for membranes and nuclear structures.
   Male-biased genes were enriched on the Y chromosome while female-biased
   genes were enriched on the X chromosome, which included X chromosome
   inactivation escapees explaining the origins of some sex differences.
   Male-biased genes were enriched for mitotic processes while
   female-biased genes were enriched for synaptic membrane and lumen.
   Finally, sex-biased genes were enriched for drug-targets and more
   female-biased genes were affected by adverse drug reactions than
   male-biased genes. In summary, by building a comprehensive resource of
   sex differences across human brain regions at gene expression level, we
   explored their likely origin and functional significance. We have also
   developed a web resource to make the entire analysis available for the
   scientific community for further exploration, available at
   [27]https://joshiapps.cbu.uib.no/SRB_app/
Supplementary Information
   The online version contains supplementary material available at
   10.1186/s13293-023-00515-w.
   Keywords: Sex difference, Human brain, Gene regulation, Hormones, Data
   integration, Conservation, Brain disorders, Drug response
Plain language summary
   Sex and gender differences are present across many organs in humans and
   have biological and social origins. The differences in brain raise a
   lot controversy due to small and sometimes contradictory results and
   its societal implications. In this study, we set out to discern the
   consistency of sex differences in brain by collecting a huge amount of
   publicly available transcriptomic data and further explore their likely
   origin and functional significance. We identified robust sex-biased
   genes in human brain with female-biased genes enriched for X chromosome
   genes. We also noted that male- and female-biased genes were enriched
   for distinct biological processes. Finally, sex-biased genes were
   enriched for androgen response elements. In summary, our analysis
   suggests sex-chromosomes and androgens as likely sources of sex
   differences in brain. Finally, we noted that age affects gene
   expression in brain more than sex.
Supplementary Information
   The online version contains supplementary material available at
   10.1186/s13293-023-00515-w.
Highlights
     * By collecting and reanalyzing 16,000 samples from 46 datasets, we
       identified robust male- and female-biased genes across 11 brain
       regions in humans. Female-biased genes were significantly enriched
       on the X chromosome and male-biased genes were enriched on the Y
       chromosome. Robust sex-biased genes were highly conserved across
       primates and other species.
     * Sex-biased genes in the brain were enriched for brain-specific
       genes, but not region-specific genes within the brain. Male-biased
       genes across brain regions were enriched for astrocyte and
       oligodendrocyte signature genes more than female-biased genes.
     * We noted that both age and sex influence gene expression for most
       genes. XIST was highly female-biased, while genes on the Y
       chromosome were male-biased. Three genes were found to be age- and
       sex-associated in both datasets, including FREM3, CHI3L1, and
       SERPINA3, which have been associated with neurological disease.
       Notably, CHI3L1 is highly expressed in female AD patients compared
       to male AD samples.
     * Over 80% of both male and female-biased genes were enriched for
       either half or full androgen response element (ARE) sites across
       brain regions, and sex-biased genes are also enriched for estrogen
       response elements (ERE).
     * Finally, we developed a web resource to make the entire analysis
       available for the scientific community for further exploration,
       available at [28]https://joshiapps.cbu.uib.no/SRB_app/
Supplementary Information
   The online version contains supplementary material available at
   10.1186/s13293-023-00515-w.
Background
   Biological sex is one of the most prominent stratification factor for
   the human population, with classical binary biological grouping into
   male and female. The physiological and behavioral sexual dimorphism in
   humans originates from both genetic and environmental constructs, and
   can produce divergent sex-specific disease susceptibility. For example,
   females carry a much higher burden of autoimmune diseases compared to
   men, while men are more likely to suffer from schizophrenia.
   Interestingly, the same alleles of the complement component 4 or C4
   genes at the major histocompatibility complex (MHC) locus were shown to
   increase risk for schizophrenia and reduce risk for autoimmune
   disorders [[29]1]. Sex and gender terms have been used inter-changeably
   in scientific literature. Sex is biologically determined by chromosomal
   makeup, while gender is more behavioral in nature and also more
   controversial as to how it is determined [[30]2]. Importantly, most
   sexually dimorphic traits are likely to be a result of multiple,
   independent sex-biasing factors where genetic and epigenetic factors
   are manifested through sex-biased gene expression or hormonal control
   [[31]3]. Such traits are defined as ’sex and gender’ or ’sex/gender
   terms’ or simply as ’sex’. Hence forth, we will use the term ’sex’ for
   simplicity.
   Male and females have many differences, in physical appearance, social
   behavior as well as in disease incidence, prevalence, morbidity and
   mortality. Yet males have been predominantly used in basic and
   pre-clinical research, due to female cyclic hormonal patterns and
   importantly, a common belief that male and females mainly have only
   reproductive difference [[32]4]. Historically clinical trials are
   largely conducted on males only and unsurprisingly, females are more
   likely to suffer from side effects from medications due to
   under-representation in clinical trials [[33]5]. Despite this,
   scientific publications in pharmacology field show a trend downward
   with 29% of articles reporting the use of both sexes in 2019 compared
   to 33% in 2009 [[34]6]. Studies of both males and females are essential
   to understanding sex-specific human biology towards the advancement of
   human health. There is growing scientific literature exploring sex
   differences in healthy lifespan and aging. Transcriptomic studies allow
   exploration of sex differences at genome-wide level providing clues for
   the molecular basis of sex differences. GTEX consortium generated
   transcriptome profiles across 53 human tissues using RNA-sequencing
   data for 544 individuals (males and females). Several studies have used
   this data to characterise sex differences across tissues
   [[35]7–[36]10]. Sex-specific differences are noted in all organs and
   these differences also affect tissues not specialized for reproduction,
   including non-reproductive tissues. Sex influences gene expression
   levels and cellular composition of tissue samples across the human
   body, with a total of 37% of all genes exhibiting sex-biased expression
   in at least one tissue [[37]10].
   Areas of the brain function differently in females and males, and are
   differentially affected by disease in the two sexes. For example, genes
   associated with Parkinson’s disease and Alzheimer’s disease are
   targeted by different sets of transcription factors in each sex
   [[38]11]. Evaluating differences in male and female brains can
   contribute to understanding sex differences in disease incidence,
   manifestation, and outcome. Accordingly, several transcriptome studies
   have focused specifically on the human brain regions to identify
   sex-biased genes [[39]12–[40]14]. Sex differences in human brain have
   nevertheless remained controversial due to small effects and
   inconsistencies across studies as most of these studies have used
   mostly one or in rare cases a few [[41]15] independent datasets making
   it hard to estimate the reproducibility of their sex-biased gene lists.
   Only a handful of studies have made a systematic effort, where the
   experimental design revealed specific causal factors for future study
   ([[42]16], Table 1). As independent validation of genes from a single
   study can be very expensive and time consuming, reproducible expression
   across studies can also be used to identify reliable sex-biased genes.
   Accordingly, we set out to investigate whether there are robust
   sex-biased gene expression signatures in human brain by collecting and
   systematically integrating vast amount of publicly available data.
   Specifically, we collected transcription profiles for more than 16,000
   samples from 46 datasets in human brain. By systematic integration of
   the data from multiple studies, we identified robust transcription
   level differences in human brain across 11 brain regions and classified
   male-biased and female-biased genes, and their likely origin and
   functional significance. We have also developed a web resource to make
   the entire analysis available for the scientific community for further
   exploration, available at [43]https://joshiapps.cbu.uib.no/SRB_app/
Methods
Data collections and differential expression analysis
   Gene expression datasets analyzed in this study were collected from
   several published brain studies (Fig. [44]1A and Additional file
   [45]1). The raw or normalised quantification matrix deposited alongside
   the original publication were re-processed and analyzed separately for
   all datasets. For the data obtained from the Gene Expression Omnibus
   (GEO) repository, the normalized gene expression were downloaded using
   the R package GEOquery 2.54.1 [[46]17]. The microarray datasets with
   raw expression values were normalized and log transformed using Robust
   Multichip Average (RMA) method. Probes without a mapping gene were
   removed. The average expression value of gene with multiple probe sets
   was calculated. Differential expression analysis were performed
   separately by 11 brain regions: amygdala (AMY), cerebellum (CBC),
   frontal lobe (FC), hippocampus (HIP), medulla and spinal cord (MED),
   occipital lobe (OC), basal ganglia (STR), temporal lobe (TC), thalamus
   (THA), parietal lobe (PC), corpus callosum (CC). The empirical Bayes
   differential expression analysis was performed by using limma 3.42.0. A
   cutoff of fold-change at 1.2 and p-value of 0.05 were used to identify
   genes as significant female-biased genes and male-biased genes. Then,
   the female- and male-biased gene lists from each dataset were ranked by
   log fold-change from the rank aggregation method. The schematic diagram
   for methods in this study is shown in Additional file [47]4: Fig. S1.
   We used the sex annotation provided by available metadata for the
   samples in all datasets. We also performed principal component analysis
   (PCA) of sex-chromosome gene expression to confirm the accuracy of
   sex-labeling of samples. Additional file [48]4: Fig. S2 shows the
   example PCA plot of Y-chromosome genes for six datasets ([49]GSE8397,
   [50]GSE12649, [51]GSE17612, [52]GSE44456, [53]GSE30483 and
   [54]GSE45642). There were very rare instances of disagreement. The
   samples were omitted from the analysis in that case.
Fig. 1.
   [55]Fig. 1
   [56]Open in a new tab
   A The number of datasets analyzed in each brain region. B A schematic
   map of the brain regions studied. C The number of female-biased (pink)
   and male-biased (blue) genes for each dataset before and after rank
   aggregation. D The number sex-biased genes across brain regions.
   E Fraction of sex-biased genes found at least in 4 primates. F Fraction
   of sex-biased genes with sex-biased genes expression at least in 2
   species. The number of genes on the sex chromosomes and autosomes in
   female-biased G and male-biased H genes
Sex-biased gene prioritization by rank aggregation method
   For each brain region, robust rank aggregation method (RRA) was used to
   combine multiple female- and male-biased rank gene lists from all
   datasets into a single prioritized female- and male-biased gene rank
   list [[57]18]. For each gene, the status was assigned as a sex-biased
   gene using combined RRA rank selected by p-value less than 0.05.
   We also applied another pipeline to define female- and male-biased
   genes. We firstly obtained differentially expressed genes by performing
   empirical Bayes differential expression analysis by using limma
   3.42.0.A. All genes in each dataset were ranked based on fold-change to
   obtain male- and female-biased gene lists. The gene lists from each
   dataset were then combined using RRA. For each brain region, combined
   gene ranking from all gene ranks into one gene rank list using RRA and
   FDR corrected P value
   [MATH: <0.05 :MATH]
   from RRA used to filter sex-biased genes, i.e., female- and male-biased
   genes (Additional file [58]4: Fig. S27).
   Correlations of sex-biased genes between brain regions were determined
   using Spearman’s correlation coefficient. Enrichment of sex-biased
   genes in X/Y-chromosomes and autosome were calculated using Fisher’s
   exact test. A conserved human gene list in six primates (’Bolivian
   squirrel monkey’, ’Chimpanzee’, ’Gorilla’, ’Gibbon’, ’Olive baboon’,
   ’Macaque’) from UCSC genome browser [[59]19] was used to investigate
   the conservation of sex-biased genes. We used SAGD database [[60]8] to
   check if sex-biased genes in human brain found in sex-biased genes of
   other species.
Gene enrichment analysis and disease-related gene analysis
   Gene Ontology (GO), Kyoto Encyclopedia of Genes and Genomes (KEGG)
   pathways significantly enriched in sex-biased genes were implemented
   using clusterprofiler (v.4.4.4) [[61]20] at adjusted p-value smaller
   than 0.05 (corrected by the Benjamini–Hochberg method). Gene–disease
   association and disease enrichment of sex-biased genes were identified
   using DisGeNet2r package [[62]21]. CURATED and all database options
   were used for disease enrichment analysis. The p-values resulting from
   the multiple Fisher’s exact tests are corrected for false discovery
   rate using the Benjamini–Hochberg method. The enrichment of Genome-Wide
   Association Studies (GWAS) Catalog 2019 were performed by enrichr
   [[63]22]. We also performed over-enrichment analysis of sex-biased
   genes in a curated brain diseased and drug-target genes from BrainBase
   database [[64]23] using Fisher’s exact test. To compare enriched terms
   across brain regions, top five significantly enriched categories of
   each brain regions were selected and plotted for visualizations for the
   enrichment. The gene count denoted by the size of the circle and adjust
   p-value denoted by the color. As enrichment analysis tools such as
   enrichr [[65]22] do not allow user-defined background genes. We also
   tested whether we observed brain-specific functional categories
   enriched in DAVID online tool [[66]24, [67]25] using all genes
   expressed in specific brain regions as a background.
Multiple regression analysis with age and sex as independent variable
   To study the contribution of age and sex only two datasets in frontal
   cortex had enough samples with a wide age range in both males and
   females. Therefore, only samples from frontal cortex brain region from
   [68]GSE11882 [[69]26] and [70]GSE53890 datasets [[71]27] were used for
   sex–age-related gene analysis. We model linear regression of each gene
   expression as linear combination of age, sex and (age*sex) variables as
   shown in Eq. 1:
   [MATH: expression=a
   ∗(age)+b∗(sex)+c∗(age∗
   sex).
   :MATH]
   Sex was created as a binary variable. Variable standardization was
   performed to reduce multicollinearity. Variance Inflation Factors (VIF)
   was used to test multicollinearity of the third independent variable
   with other independent variables. The cutoff of regression coefficient
   of age and sex variables were used to identify age- or sex-related
   genes (Additional file [72]4: Fig. S3).
Cell-type and tissue-specific enrichment analysis
   Enrichment of our sex-biased genes in two cell type-specific gene lists
   was calculated. First set of cell type-associated genes was from
   McKenzie et al. [[73]28]. Fisher’s exact test was used to test for cell
   type-specific tissue. The second cell-type gene list was from Dougherty
   et al. [[74]29]. In this section, specific expression analysis across
   cell types (CSEA) web tool was used to calculate Fisher’s exact test
   with Benjamini–Hochberg correction of the overlap between our
   sex-biased genes and their cell type-specific genes. In order to
   investigate whether sex-biased genes are highly enriched or specific
   expression in brain. Tissue-specific enrichment analysis (TSEA) was
   performed using deTS package with GTEx panel [[75]30].
Androgen response element (ARE), estrogen response element (ERE) and motif
analysis
   In order to determine the number of androgen (AR) and estrogen
   receptors (ER) in sex-biased genes, genes with full and half ARE and
   ERE binding sites from published studies [[76]31, [77]32] were used to
   find an overlap genes between these receptor genes and sex-biased,
   brain expressed and brain regionally elevated genes (from the human
   protein atlas). Known motif enrichment analysis in the promoters of
   sex-biased genes was performed by HOMER (v4.11) [[78]33].
Drug–target interactions and adverse drug response
   We used drug–target interactions with 2118 drugs/chemicals from
   BrainBase database [[79]23]. Fisher’s exact test was used to calculate
   over-enrichment for drugs target in sex-biased genes. The enrich terms
   with p-value less than 0.0001 were plotted across all regions. The
   adverse drug reaction genes from Chen et al. [[80]34] was also used to
   calculated overlap with sex-biased genes.
Results
Sex-biased gene expression across 11 human brain regions
   Many studies have been published to identify sex-biased genes in one or
   several brain regions, but assessment of the robustness of these
   sex-biased genes is missing. In order to identify a robust sex-biased
   gene signature, i.e., sex-biased genes supported by multiple studies,
   we collected over 16,000 individual samples from 46 gene expression
   datasets in human brain (Fig. [81]1A). The samples were grouped into 11
   brain regions namely, amygdala (AMY), cerebellum (CBC), frontal lobe
   (FC), hippocampus (HIP), medulla and spinal cord (MED), occipital lobe
   (OC), basal ganglia (STR), temporal lobe (TC), thalamus (THA), parietal
   lobe (PC), and corpus callosum (CC) (Fig. [82]1B). Individual datasets
   consisted diverse human sample material, experimental protocols and
   technology. We selected datasets with genome-wide expression data
   generated using either microarray or RNA sequencing technologies and
   with a minimum of ten samples of each sex. Technical and technological
   divergence across datasets, complicated pooling of samples. We
   therefore identified male-biased and female-biased genes in each
   individual dataset using differential expression analysis (p-value
   [MATH: <0.05 :MATH]
   and 1.2 or more fold-change) and further used robust rank aggregation
   method [[83]18] to combine multiple ranked lists of sex-biased genes
   from different datasets (Additional file [84]4: Fig. S1), resulting
   into a robust male and female-biased gene list in each brain region.
   Fig. [85]1C (left box) shows the number of sex-biased genes (male—blue,
   female—pink) in each individual dataset, whereas Fig. [86]1C (right
   box) represents the number of sex-biased genes after the rank
   aggregation in the FC brain region (Additional file [87]4: Figs. S4 and
   S5 and Additional file [88]2 for female-biased genes and Addition file
   [89]3 for male-biased genes). We noted that there were only a handful
   of genes (
   [MATH: <5 :MATH]
   ) detected as sex-biased across all studies in each brain region. This
   is likely due to heterogeneity of data caused by multiple factors.
   First and foremost, the heterogeneity across the human samples with
   diverse demographic and socioeconomic traits as well as the
   technological heterogeneity in the data including multiple platforms,
   different experiment protocols, and unequal sample size. Most of our
   sex-biased genes were identified as sex-biased in at least two
   datasets. There were on average about a hundred sex-biased genes in
   each of the 11 brain regions. THA, TC and FC with the most sex-biased
   genes while AMY and CC had the lowest number of sex-biased genes. There
   was no correlation between the number of sex-biased genes and the
   number of available datasets or the total number of samples across
   regions (Additional file [90]4: Fig. S6). Both male- and female-biased
   genes were present across all brain regions with a very small bias for
   male-biased genes than female-biased genes (Fig. [91]1D).
   To estimate the conservation of sex-biased genes, we downloaded a list
   of conserved human genes in six primates (‘Bolivian squirrel monkey’,
   ‘Chimpanzee’, ‘Gorilla’, ‘Gibbon’, ‘Olive baboon’, ‘Macaque’) from UCSC
   genome browser [[92]19]. About 80% of both male and female-biased genes
   were found in at least four primates compared to only about half of all
   human genes (black) conserved in at least four primates (Fig. [93]1E).
   Sex-biased genes in nearly all brain regions were therefore highly
   conserved across primates. We also estimated the conservation of
   sex-biased genes in higher eukaryotes from Ensembl. Lists of conserved
   human genes in 198 species were downloaded from Ensembl genome browser.
   Both male and female-biased genes were more conserved across other
   species than all genes (Additional file [94]4: Fig. S7). In order to
   check whether human sex-biased genes in brain show sex-biased
   expression in other species, we used SAGD database [[95]8]. The SAGD
   database consists of sex-associated genes across organs in diverse
   species. Our human brain sex-biased genes were indeed enriched for
   sex-biased genes in other species. The fraction of male and
   female-biased with sex-associated genes in at least two species
   (excluding human) was significantly higher than all human genes (Fig.
   [96]1F).
   We further checked whether sex-biased genes were enriched in specific
   genomic regions. Female-biased genes were significantly enriched on the
   X chromosome (Fig. [97]1G) and male-biased genes were enriched on the Y
   chromosome (Fig. [98]1H) as expected. For example, X chromosome
   contains about 5% of human genes and about 20% of female-biased genes
   in medulla were on the X chromosome. Similarly, Y chromosome contains
   about 1% of human genes and about 40% of male-biased genes in amygdala
   were on the Y chromosome. Furthermore, we checked whether the genes on
   sex chromosomes belonged to the pseudoautosomal regions (PAR1 and PAR2)
   of the human X and Y chromosomes which do recombine during meiosis. We
   noted that male-biased genes on Y chromosome and female-biased genes on
   X chromosome were not enriched for genes on pseudoautosomal regions
   (Additional file [99]4: Fig. S9A). XIST, a long non-coding RNA
   expressed from X chromosome ensures that one of the pair of X
   chromosomes is transcriptionally silenced (X chromosome inactivation or
   XCI) during early development in mammalian females. Many genes on X
   chromosome evade this dosage equivalence providing a mechanism for
   divergence between males and females, called XCI escape genes
   [[100]35]. The female-biased genes on X chromosome highly overlapped
   with XCI escape genes (Additional file [101]4: Fig. S10B). In summary,
   we noted that sex-biased genes were enriched for sex chromosomes and
   were located on the sex-specific part of each chromosome. We noted no
   preference for autosomes for both male and female sex-biased genes.
   To explore the functional relevance of sex-biased genes, we first
   conducted the pathway enrichment analysis using Gene Ontology (GO) and
   Kyoto Encyclopedia of Genes and Genomes pathway annotations (Fig.
   [102]2, bigger figure shown in Additional file [103]4: Figs. S11–S14).
   The functional enrichment of female-biased genes was brain region
   specific with CBC and FC genes enriched for zinc and copper response
   while OC genes for neuronal activity (Fig. [104]2A, B and Additional
   file [105]4: Fig. S11). In cellular component enrichment analysis, the
   most significant enrichment terms of female-biased genes were related
   to postsynaptic and synaptic membrane in OC and PL and lumen in FC and
   HIP (Fig. [106]2C and Additional file [107]4: Fig. S12A). While
   male-biased genes were related to DNA packaging, spindle and nucleosome
   in many brain regions (Fig. [108]2D and Additional file [109]4: Fig.
   S12B). In summary, female-biased genes were enriched for
   neuron-associated processes while male-biased genes were enriched for
   nuclear structures. Enrichment of Disease-Related Genes in sex-biased
   genes across brain regions were examined using BrainBase, DisGeNet
   (curated) and GWAS catalog 2019 database (Fig. [110]2E, F, and
   Additional file [111]4: Figs. S15, 16). Sex-biased genes were highly
   enriched for genes related to glioma across many brain regions (Fig.
   [112]2E, F). Alzheimer’s related genes were highly enriched for the
   female-biased genes across four brain regions (Fig. [113]2E). SFARI
   database ([114]https://gene.sfari.org/) [[115]36] contains about 1000
   genes related to autism spectrum disorders (ASD). Female-biased genes
   in several brain regions were enriched for ASD-related genes
   (Additional file [116]4: Fig. S17).
Fig. 2 .
   [117]Fig. 2
   [118]Open in a new tab
   The top 5 enriched terms for the gene ontology and disease enrichment
   analysis across brain regions. A Biological process enrichment for
   female-biased genes. B Biological process enrichment for male-biased
   genes. C Cellular component enrichment for female-biased genes
   D Cellular component enrichment for male-biased genes. E BrainBase
   disease enrichment analysis for female-biased genes. F BrainBase
   disease enrichment analysis for male-biased genes
Sex-biased gene overlap across brain regions
   Given that functional enrichment showed high overlap across male-biased
   genes, we hypothesized high overlap among male-biased gene sets
   compared to female-biased gene sets across brain regions. Indeed, the
   overlap of sex-biased genes showed that male-biased genes were more
   shared with 14 genes were male-biased in all 11 brain regions (Fig.
   [119]3B) compared to only three genes were female-biased in all brain
   regions (Fig. [120]3A). Importantly, female- and male-biased genes
   found in more than eight brain regions were located on X- and
   Y-chromosomes, respectively.
Fig. 3.
   [121]Fig. 3
   [122]Open in a new tab
   A The number of female-biased genes by number of regions. The color
   grey, red and blue are shown bar graphs for proportions of genes mapped
   into autosome, X-chromosome and Y-chromosome, respectively. B The
   number of male-biased genes by number of regions. C The correlation
   heatmap of female-biased genes. D The correlation heatmap of
   male-biased genes. E DisGeNet (CURATED) enrichment of overlap
   female-biased genes across FC, PL, TC and OC F DisGeNet (CURATED)
   enrichment for overlap female-biased genes across FC, PL, TC and OC
   We further calculated a pair-wise overlap of male and female-biased
   genes across brain regions. Female-biased genes in each brain region
   showed very little overlap with other brain regions (Fig. [123]3C),
   while the male-biased genes grouped the brain regions in two core
   clusters (Fig. [124]3D). Male-biased genes in AMY, CC and MED showed a
   high overlap. We noted that sex-biased genes in PL, FC, TC and OC
   showed a distinct signature in both males and females with a high
   correlation between these brain regions (Fig. [125]3C, D). Overlap
   sex-biased gene lists of PL, FC, TC and OC were further examined for
   gene-disease association enrichment from databases GWAS catalog 2019
   and DisGeNet (curated database) (Fig. [126]3E, F and Additional file
   [127]4: Fig. S18). The female-biased genes in four regions were
   enriched for Alzheimer’s disease progression (SYN3 and STK32B) and the
   male-biased genes in four brain regions were enriched for neuroticism
   (PAX6 and PLTP). The DisGeNet enrichment for female-biased genes in
   four brain regions identified many mental disorders (Fig. [128]3E,
   Additional file [129]4: Fig. S19A).
Cell-type and tissue specificity of sex-biased genes
   So far, we identified robust sex-biased genes and noted that
   male-biased genes across brain regions showed higher overlap in the
   previous sections. To check whether sex-biased genes show
   brain-specific gene expression, we performed tissue enrichment analysis
   using deTS [[130]30]. The overlap of tissue-specific genes and
   sex-biased genes revealed that both male and female-biased genes
   significantly overlapped with brain-specific genes. Specifically, in
   almost all brain regions, brain tissues were the only enriched tissues
   out of a total of 48 body tissues for sex-biased genes (Additional file
   [131]4: Fig. S20). However, the sex-biased genes were not enriched for
   the genes specific to the individual brain regions, i.e., sex-biased
   genes in hippocampus did not show highest enrichment for deTS
   hippocampus genes. In summary, we observed that the sex-biased genes
   were brain specific compared to other body tissues but not brain region
   specific within the brain (Additional file [132]4: Fig. S20).
   Given that sex-biased genes were enriched for brain-specific genes, we
   further explored whether there was a cell-type specificity for
   sex-biased genes in brain. We used brain cell signature gene lists for
   five cell types (astrocytes, oligodendrocytes, microglia, neurons and
   endothelial cells) from McKenzie et al. gene sets [[133]28] and
   calculated significant overlap using different thresholds for both male
   and female-biased genes (see "[134]Methods"). As expected, both male
   and female-biased genes were enriched for many cell-type signature
   genes across brain regions (Fig. [135]4A, B bigger figure shown in
   Additional file [136]4: Fig. S21)). We noted that male-biased genes
   across brain regions were enriched for astrocytes and oligodendrocyte
   signature genes more than female-biased genes. A previous study
   exploring genes exhibiting sex-biased expression in human fetal brain,
   noted that the male-biased genes were enriched for expression in neural
   progenitor cells, whereas female-biased genes are enriched for
   expression in Cajal–Retzius cells and glia [[137]37]. This observation
   was not supported in our analysis of adult brain regions. We also used
   an independent resource of cell type-specific expression in human brain
   [[138]38] to calculate cell type enrichment of male and female-biased
   genes (Additional file [139]4: Fig. S22–S25). Indeed, male-biased gene
   enrichment for astrocytes and oligodendrocytes in FC, OC, PL and TC is
   supported by both the datasets (Fig. [140]4B).
Fig. 4.
   [141]Fig. 4
   [142]Open in a new tab
   A Cell-type enrichment for female-biased genes. B Cell-type enrichment
   for male-biased genes. C Transcription factor (TF) enrichment for
   female-biased genes. D Transcription factor (TF) for male-biased genes.
   E The percentage of overlap between androgen receptor element (ARE)
   genes and sex-biased genes, brain expressed genes, and brain regionally
   elevated genes. The colors grey, red, yellow and orange in bar graphs
   represent the proportion of genes that not overlap, overlap with ARE
   full sites genes, overlap with ARE half sites genes and overlap both in
   ARE full and half sites, respectively. F The percentage of overlap
   between estrogen receptor element (ERE) genes and sex-biased genes,
   brain expressed genes and brain regionally elevated genes. The color of
   grey and peach are shown the proportion of genes that not overlap and
   overlap with ERE genes, respectively
Regulatory mechanisms behind sex-biased gene expression
   To explore possible transcription regulatory mechanisms behind
   sex-biased genes, we firstly performed known motif enrichment analysis
   in the promoters of the sex-biased genes. The analysis did not identify
   strong enrichment for motifs of specific transcription factors
   (Additional file [143]4: Fig. S18) for both male and female-biased gene
   sets. We then obtained a reconstructed transcription regulatory network
   model in human brain by integrating brain-specific DNase footprinting
   and TF-gene co-expression [[144]39]. This network consisted of over 700
   transcription factor and their predicted targets. The enrichment
   analysis of predicted transcription factor targets in male and
   female-biased gene list identified many potential transcription factors
   (Fig. [145]4C, D). Male-biased genes in TC, FC, OC and PL had a high
   overlap. Accordingly, many transcription factors, notably SOX family
   member targets were enriched in these four regions in male-biased genes
   (Fig. [146]4D). SOX2 and SOX9 putative targets highly overlapped with
   female-biased genes in two brain regions (HIP and THA) (Fig. [147]5C).
   Interestingly, female-biased genes in HIP and THA had nearly no overlap
   (Fig. [148]3C).
Fig. 5.
   [149]Fig. 5
   [150]Open in a new tab
   A The scatter plot of the coefficients of age and gender variables from
   the multiple linear regression from [151]GSE11882. B The scatter plot
   of the coefficients of age and gender variables from the multiple
   linear regression from [152]GSE53890 dataset. C Gene expression of XIST
   gene in [153]GSE53890 dataset, labeled as red and blue for female and
   male samples, respectively. D Gene expression of RPS4Y1 genes in
   [154]GSE53890 dataset, colored red and blue for female and male
   samples, respectively. E Gene expression of CALB1 genes in
   [155]GSE53890 dataset, colored red and blue for female and male
   samples, respectively. F Gene expression of FKBP5 genes in
   [156]GSE53890 dataset, colored red and blue for female and male
   samples, respectively. G Gene expression of FKBP5 genes in
   [157]GSE53890 dataset, colored red and blue for female and male
   samples, respectively. H Venn diagram of overlap genes between
   sex-biased genes and age-biased genes from [158]GSE53890 and
   [159]GSE11882 datasets
   Sex-specific hormones can mediate sex-biased gene expression. We
   therefore obtained genes enriched for the hormone response elements.
   The overlap between our robust sex-biased genes and androgen response
   elements (ARE) and estrogen response elements (ERE) was calculated. We
   noted that over 80% of both male and female-biased genes were enriched
   for either half or full ARE sites across brain regions (Fig. [160]4E).
   This fraction is significantly higher than all human genes with about
   50% genes with ARE half or full sites (Fig. [161]4E). We also obtained
   gene lists with highly expressed genes in specific brain regions called
   regionally elevated genes from Allan Brain Atlas. This genes showed
   similar enrichment to sex-biased genes for ARE half and full sites
   (Fig. [162]4E). The analysis of ERE binding sites provided with results
   similar to ARE binding sites, i.e., sex-biased and regionally elevated
   genes were enriched for ERE sites compared to all genes (Fig. [163]4F).
   In summary, sex-biased genes are enriched for sex hormone response
   elements.
Age and sex relationship in brain gene expression
   We previously noted that one of the likely reasons for the low overlap
   in sex-biased genes across different studies is the fact that brain
   samples came from very diverse human cohorts with heterogeneity in many
   socio-demographic traits including age. To dissect, sex and age
   components, we selected datasets covering samples in a wide age range
   for both sexes. Only two datasets from the human frontal cortex
   provided sample variability in age to allow estimation of age and sex
   effect on the gene expression. We therefore evaluated the effect the
   sex and age on brain gene expression using two datasets; [164]GSE11882
   [[165]26] and [166]GSE53890 [[167]27]. Multiple linear regression for
   individual genes was performed using age, sex and age*sex as
   independent terms in each dataset ([168]Methods for details). The
   coefficients for the age and sex terms were used to select sex-biased
   and age-biased genes (Fig. [169]4A, B). The regression coefficient for
   age*sex term for about 50% genes was greater than individual age or sex
   variable, demonstrating that both age and sex influence gene expression
   for most genes.
   We noted that XIST was highly sex specific and female biased (Fig.
   [170]5C) and genes on Y chromosome—ribosomal protein S4 (RPS4Y1),
   KDM5D, USP9Y and DDX3Y were male-biased, as expected (Fig. [171]5D). On
   the other hand, some genes showed expression variability mainly through
   aging. For example, calcium binding protein, CALB1 decreased during
   aging (Fig. [172]5E), while immune regulatory gene FKBP5 increased
   during aging (Fig. [173]5F) consistently in both males and females. We
   noted that many female-biased genes decreased gene expression during
   aging while many male-biased genes increased in gene expression. For
   example, Cluster of differentiation 99 (CD99) expression was
   male-biased and increased during aging (Fig. [174]5G). We identified
   age and sex-associated genes in each dataset (see Methods) and
   calculated overlap between them (Fig. [175]5H). Only 8 genes were sex
   associated in both datasets (FREM3, DDX3Y, KDM5D, SERPINA, USP9Y, XIST,
   CHI3L1, EIF1AY) and fourteen genes were age associated in both datasets
   (CBLN4, FREM3, AKAP5, C11orf87, CRH, LINC00507, SERPINA3, CALB1, RGS4,
   CHI3L1, AQP1, VIP, S100A8, NETO2). Age and sex-associated genes had a
   high overlap in each dataset (Fig. [176]5H) and three genes were found
   age- and sex- associated in both datasets. FREM3 was female-biased
   while CHI3L1 and SERPINA3 genes were male-biased, and the expression of
   CHI3L1 and SERPINA3 increased with age while the expression of FREM3
   genes decreased with age. All these genes have been shown to be
   associated with neurological disease [[177]40–[178]42]. FREM3 is
   associated with depression and aging in human brain [[179]40]. Another
   study also found sex-, age- and Alzheimer’s disease-related differences
   in CHI3L1 expression in the brain. Interestingly, CHI3L1 is highly
   expressed in female AD patients compared to male AD samples [[180]41].
Sex-biased drug response
   After evaluating the likely regulatory factors of sex-biased genes, we
   explored the clinical impact of sex-biased expression. It is well
   documented that males and females have differential response to many
   drugs. We used drug–target interactions covering 2118 drugs or
   chemicals and 623 genes from BrainBase database [[181]23] to calculate
   the enrichment for drugs in sex-biased genes. Many drug targets were
   enriched for sex-biased genes (P value
   [MATH: <0.0001 :MATH]
   ) in both males and females, particularly in FC, PL and TC brain
   regions. More female-biased genes overlapped with drug targets than
   male-biased genes. For example, midazolam target genes were
   female-biased in FC and aspirin targets were male-biased in TH. Indeed,
   midazolam, a sedative and anesthetic adjuvant, has demonstrated
   sex-specific effects with deeper sedation in men compared with women
   [[182]43] and sex difference in aspirin response is also well known
   where women are 2.5 times more likely to be aspirin resistant than men
   [[183]44]. Cisplatin targets were enriched in male-biased genes in
   temporal and occipital CC. Cisplatin-related gender differences in
   nephrotoxicity also showed greater damage in males than females
   [[184]45]. Antipsychotic and antidepressant targets were enriched for
   female-biased genes. There are known sex differences in pharmacodynamic
   effects of many drugs. In women, they include greater sensitivity to
   and enhanced effectiveness of beta blockers, opioids, selective
   serotonin reuptake inhibitors, and typical antipsychotics.
   Additionally, women are 50–75% more likely than men to experience an
   adverse drug reaction [[185]46]. We therefore further explored whether
   the genes associated with adverse drug reactions were sex-biased. We
   obtained adverse drug reaction genes from Chen et al. [[186]34] and
   calculated overlap with sex-biased genes. Both male and female-biased
   genes overlapped with many adverse drug reaction phenotypes
   (Fig. [187]6C, D, y axis). However, female-biased genes showed a higher
   overlap of genes for most of adverse drug reaction phenotype
   (Fig. [188]6C).
Fig. 6.
   [189]Fig. 6
   [190]Open in a new tab
   A Over-enrichment of BrainBase drug targets for female-biased genes.
   B Over-enrichment of BrainBase drug targets for male-biased genes.
   C Number of genes overlapping between female-biased genes and adverse
   drug reaction genes (left). Number of genes overlapping between
   male-biased genes and adverse drug reaction genes (right)
A web resource of sex-biased gene expression analysis in human brain
   We developed a publicly available web resource to provide access to the
   key analysis of sex-biased genes. SexRankBrain is an R shiny
   interactive tool [[191]47] to explore the sex-biased genes across
   datasets from human brain. This web resource, in addition also allows
   the robustness analysis of our findings as it allows users to change
   different thresholds during the analysis. We utilized this feature of
   the web resource to confirm that major finding noted in this study were
   consistent at different thresholds. Users can set thresholds to obtain
   sex-biased gene lists from all datasets for each brain regions in this
   study. These lists can then be used in the web application for
   calculating sex-biased gene rank using custom parameter from user and
   create a result dashboard. There are three module tabs in the
   application. The first and second tab allow users to explore the
   functional features of sex-biased genes for individual brain regions as
   well as a comparison of sex-biased genes across all brain regions,
   respectively. The third tab contains information about web-application.
   In the first tab, the web-app allows user to select their preference
   cutoff for a specific brain region in three steps. First step is to
   apply p-value and logFC cutoff for sex-biased genes filtering in all
   datasets. Next, the web application performs a robust rank aggregation
   (RRA) from all gene ranks and creates a combined sex-biased gene rank
   [[192]18] for each brain region. Users can choose a custom RRA p-value
   cutoff to filter significant sex-biased genes from an aggregate rank.
   The last step is to perform diverse enrichment analyses of significant
   sex-biased genes. Gene ontology (GO), Kyoto Encyclopedia of Genes and
   Genomes (KEGG) pathways and DisGeNet [[193]21] enriched in sex-biased
   genes are implemented using Enrichr [[194]22]. The web application has
   a second tab for the comparison of sex-biased genes across brain
   regions. Here, users can similarly choose parameters for the steps as
   described in the first tab. The enrichment results for this tab are
   implemented by compareCluster() function in clusterProfiler package
   [[195]20]. For both tabs, the tables of custom filtered rank genes and
   all individual figures can be downloaded. The open source code for the
   shiny application is available on GitHub
   ([196]https://github.com/PattaWapee/SexRankBrain).
Discussion
   Males and females display a plethora of divergent physical and
   behavioral patterns, affecting many life outcomes including disease
   prevalence, symptoms, and progression rates. Funding agencies and
   publishers are calling for greater attention to exploring basic
   biological processes and disease mechanisms across the sexes and
   genders. Besides the reproductive organs, most sex differences in the
   body are quantitative, i.e., the distribution differ between the two
   sexes but largely overlaps, as is the case with the height or the brain
   volume, as well as many other physiological traits such as stress,
   opioid sensitivity, and immune response [[197]16]. Accordingly, studies
   exploring sex differences during development and disease in humans have
   exploded in recent years. Nevertheless, most of them derived their
   conclusions based on only single or a handful of datasets
   [[198]7–[199]10]. A recent study [[200]10] noted sex differences across
   many cell types in humans including brain, albeit only using one
   dataset, unlike this study. The study [[201]10] did not focus
   specifically on the brain but many of our findings were indeed
   overlapping, e.g., enrichment of sex-biased genes on sex chromosomes.
   The sex differences in brain are controversial mainly because of its
   likely societal implications. Women have been culturally oppressed and
   sex differences have been used as a justification for some of the
   injustices. Nevertheless, the sex differences in the brain may explain
   the differences seen in prevalence, symptomology and even treatment for
   brain pathologies. It is therefore important to establish the validity
   of observed sex differences across different studies. We therefore
   combined a huge amount publicly available expression data to estimate
   sex differences in human brain. Firstly and most importantly, we noted
   that most of sex-biased genes obtained from one data were not
   identified in other datasets. This is partially due to heterogeneity in
   data, e.g., the impact of the age of individuals (explored in detail in
   this paper), as well as other confounders, and technical and
   technological differences across studies. By systematic data
   integration, we obtained a robust sex-biased gene list for each of 11
   brain regions. The robust sex-biased genes were highly conserved and
   showed sex-biased gene expression in other species as well. This
   allowed us to validate some of the findings obtained previously using
   individual datasets as well as generated some novel hypotheses. We
   first established that our findings are not sensitive to a specific
   threshold of the analysis pipeline (analysis available as a web
   resource). Furthermore, we used another independent pipeline to define
   sex-biased genes (see [202]Methods for details and Additional file
   [203]4: Fig. S27). Using this alternative pipeline (Additional file
   [204]4: Fig. S27), we validated that main findings described in this
   manuscript are not dependent on the specifics of the analysis pipeline
   (Additional file [205]4: Figs. S27–30), providing additional confidence
   in the findings.
   Arnold proposed a general theory of mammalian sexual differentiation
   whereby sex chromosome genes are the primary factors causing sexual
   differentiation [[206]3]. The biggest genetic distinguishing factor
   between two sexes is the presence of sex chromosome where x chromosome
   causes sex differences in gene expression through XIST, X genes
   escaping inactivation, and imprinted X genes [[207]3]. For example,
   Kassam et al. [[208]9] observed that X-linked KAL1 gene had higher
   expression in females than males in lung tissue. The biallelic
   expression of KAL1 gene in lung tissue is an example of tissue-specific
   escape from X-activation [[209]48]. Indeed, the sex-biased genes in
   females were enriched on the X chromosome and particularly for the XCI
   escapee genes. This suggests that a part of sex-biased gene expression
   originates from the XCI escape mechanisms. Also, male-biased genes in
   brain were enriched for Y chromosome. On the other hand, it was noted
   that 90% of sex-biased genes across human tissues were mapped to
   autosomes, thus it’s not restricted within sex chromosomes [[210]15].
   This finding was partly supported in our study where most sex-biased
   genes in all brain regions were expressed from autosomes, rather than
   sex chromosomes. It is important to note that our robust sex-biased
   genes contained a higher fraction of genes (15–40%) on sex chromosomes,
   i.e., genes on sex chromosomes are more likely to be validated across
   studies and across multiple tissues. Furthermore, sex-biased genes
   found in more than eight brain regions were primarily on the sex
   chromosomes. X chromosome is particularly enriched for genes involved
   in brain-related functions. Many functional enrichment analysis tool
   including enrichr do not allow user-defined background genes. We
   therefore validated the brain-related functional enrichment in the
   sex-biased genes using DAVID online tool [[211]24, [212]25] by
   providing brain region specific background genes (Additional file
   [213]4: Fig. S31).
   Sex chromosomes are thought to regulate gene expression manifesting sex
   differences in brain primarily through the steroid hormones.
   Accordingly, we noted that both male and female-biased genes were
   enriched for both androgen and estrogen receptor binding sites. These
   findings departs from the traditional model of testosterone
   masculinizes the brain of males away from a default female form and
   supports a model where sex effects on the brain of both females and
   males are exerted by genetic, hormonal, and environmental factors.
   These factors act via multiple partly independent mechanisms that may
   vary according to internal and external factors [[214]49].
   We further tested the specific cell type enrichment for the robust
   sex-biased genes. Our results are in somewhat agreement with the
   previous reports that non-neuronal cells and inflammatory mediators
   were found in greater number and at higher levels in male brains
   [[215]50]. The higher baseline of inflammation is speculated to
   increase male vulnerability to developmental neuropsychiatric disorders
   that are triggered by inflammation [[216]50]. We noted a strong male
   bias for astrocytes and oligodendrocytes but not microglia.
   Nevertheless it is important to note that, gene expression is affected
   many factors. Kang et al. [[217]51] studied the spatio-temporal
   dynamics of the human brain transcriptome to note that age contributed
   more to the global differences in gene expression than sex. For
   example, in middle-aged women, the gene expression changes were higher
   for astrocytes, endotheliocytes, and microglia compared to young women
   [[218]52]. We performed a systematic analysis of the two datasets to
   estimate the effect of age and sex on the gene expression to note that
   for most genes sex and age both influence expression. Some age-related
   traits are conserved across sexes, there is age-related activation of
   immune- and inflammation-related genes in both male and female brains
   [[219]26], while others are affected by both sex and age. Males showed
   significantly more gene expression changes in brain through aging with
   substantial gene change in the transition to the sixth and seventh
   decades of life. In contrast, females showed the largest numbers of
   genes responding in the eighth and ninth decades of life [[220]26].
   Schizophrenia has a more severe course (negative symptoms as well as
   cognitive impairment), experienced earlier in life in boys than in
   girls [[221]53]. We explored the clinical implications the sex
   differences and noted that female-biased genes showed a high overlap
   with Alzheimer-related genes. Importantly, we also noted that more
   female-biased genes are involved in adverse drug reactions. Despite
   funders (e.g. NIH) pushing for female inclusion in clinical studies,
   very few (less than 10 percent of studies) are examining health issues
   related to females [[222]54].
Perspectives and significance
   In summary, by integrating large amount of expression data, we
   identified robust sex differences across human brain regions. We have
   made entire analysis available as a web resource at
   [223]https://joshiapps.cbu.uib.no/SRB_app/ for further exploration and
   hypothesis generation. Sex, together with age and other factors,
   affects brain function through human life span. Heterogeneity of human
   samples in many gene expression cohorts therefore makes it challenging
   to discern exactly the sex component. This indeed is a major
   shortcoming of many studies including this one. The finding of this
   study emphasized the importance of the need for greater attention to
   exploring basic biological processes and disease mechanisms in a sex
   and gender context. This study provided a foundation for future
   research to further investigate the mechanisms and factors contributing
   to sex and gender differences in the human brain.
Supplementary Information
   [224]13293_2023_515_MOESM1_ESM.xlsx^ (14.4KB, xlsx)
   Additional file 1. List of published datasets used in this study.
   [225]13293_2023_515_MOESM2_ESM.xlsx^ (86.2KB, xlsx)
   Additional file 2. The table of female-biased gene in each brain
   regions with chromosome information and check-tick in all datasets used
   in this study.
   [226]13293_2023_515_MOESM3_ESM.xlsx^ (105.6KB, xlsx)
   Additional file 3. The table of male-biased gene in each brain regions
   with chromosome information and check-tick in all datasets used in this
   study.
   [227]13293_2023_515_MOESM4_ESM.pdf^ (22.9MB, pdf)
   Additional file 4: Figure S1. Schematic diagram of the workflow
   including rank aggregation for sex-biased genes detection in this
   study. Figure S2. Principal component analysisplot of Y- chromosome
   genes expression for GSE8397, GSE12649, GSE17612, GSE44456, GSE30483
   and GSE45642. Figure S3. The cutoff for coefficient of age and sex
   variables in section multiple regression analysis with age and sex as
   independent variable. Figure S4. The number of sex-biased genes in
   individual datasets, aggregated lists in AMY, CBC, CC, HIP, MED and OC.
   Figure S5. The number of sex-biased genes in individual datasets,
   aggregated lists in PL, STR, TC and THA. Figure S6. Plot of number of
   sex-biased genes in different number of brain samples. Figure S7.
   Fraction of sex-biased genes found at least in 40 species and 160
   species. Figure S8. The number of overlapping sex-biased genes by
   number of datasets for AMY, CBC, CC, FC, HIP and MED. Figure S9. The
   number of overlapping sex-biased genes by number of datasets for OC,
   PL, STR, TC and THA. Figure S10. The number of par1 genes in
   male-biased genes and the number of XCI female-biased genes. Figure
   S11. Gene ontology enrichment analysis of sex-biased genes for
   biological process. The top 5 enrich terms across brain regions in
   female-biased genes and male-biased genes. Figure S12. Gene ontology
   enrichment analysis of sex-biased genes for cellular component. The top
   5 enrich terms across brain regions in female-biased genes and
   male-biased genes. Figure S13. Gene ontology enrichment analysis of
   sex-biased genes for molecular functions. The top 5 enrich GO terms
   across brain regions in female-biased genes and male-biased genes.
   Figure S14. KEGG pathway enrichment analysis of sex-biased genes. The
   top 5 enrich GO terms across brain regions in female-biased genes and
   male-biased genes.Figure S15. GWAS catalog 2019 enrichment across brain
   regions in female-biased genes and male-biased genes. Figure S16.
   DisGeNET enrichment analysis of sex-biased genes across brain regions
   in female-biased genes and male-biased genes. Figure S17. The number of
   autism genes found in sex-biased genes. Autism genes come from SFARI
   database. Figure S18. DisGeNetenrichment of overlap sex-biased genes
   across FC,PL,TC and OC in female-biased genes and male-biasd genes.
   GWAS catalog 2019 enrichment analysis of overlap sex-biased genes
   across FC, PL, TC and OC in female-biased genes and male-biased genes.
   Figure S19. Gene-disease class heatmap of DisGeNET for overlap
   sex-biased gene lists of PL, FC, TC and OC. Diseases are grouped by the
   their MeSH disease classes. The color scale is related to the
   percentage of disease in each class. Figure S20. Tissue-specific
   enrichment analysisof female-biased genes and male-biased genes. Figure
   S21. Cell type enrichment of sex-biased genes from McKenzie et al gene
   sets. Figure S22. Cell type specific expression analysisof female
   biased genes in AMY, CBC, CC, FC, HIP and PL. Figure S23. Cell type
   specific expression analysisof female biased genes in STR, TC and THA.
   Figure S24. Cell type specific expression analysisof male biased genes
   in CBC, CC, FC, HIP, MED and OC. Figure S25. Cell type specific
   expression analysisof male biased genes in PL, STR, TC and THA. Figure
   S26. Known motif enrichment analysis by HOMER in the promoters of the
   female-biased genes and male-biased genes. Figure S27.Schematic diagram
   of additional workflow to generate sex-biased gene list.The number of
   sex-biased genes from the additional workflow. Figure S28.The number of
   female-biased genes across brain regions using additional workflowThe
   number of male-biased genes across brain regions using additional
   workflow.The correlation heatmap of female-biased genes using
   additional workflow.The correlation heatmap of male-biased genes using
   additional workflow.The percentage of overlap between Androgen receptor
   elementgenes and sex-biased genes, brain expressed genes, and brain
   regionally elevated genes using additional workflow.The percentage of
   overlap between Estrogen receptor elementgenes and sex-biased genes.
   brain expressed genes and brain regionally elevated genes using
   additional workflow. Figure S29.Fraction of sex-biased genes found at
   least in 40 primates using additional workflow.Fraction of sex-biased
   genes found at least in 160 primates using additional workflow.Gene
   ontology enrichment analysis of female-biased genes for biological
   process using additional workflow.Gene ontology enrichment analysis of
   male-biased genes for biological process using additional workflow.Gene
   ontology enrichment analysis of female-biased genes for molecular
   function using additional workflow.Gene ontology enrichment analysis of
   male- biased genes for molecular function using additional workflow.
   Figure S30.Gene ontology enrichment analysis of female-biased genes for
   cellular composition using additional workflow.Gene ontology enrichment
   analysis of male-biased genes for molecular cellular composition using
   additional workflow.DisGeNetenrichment of female-biased genes using
   additional workflow.) DisGeNetenrichment of male- biased genes using
   additional workflow. Figure S31. Gene ontology analysis of output from
   DAVIDfor biological process.The top GO terms in female-biased genes in
   THA region with background genes of THA gene expression data.The top GO
   terms in male- biased genes in THA region with background genes of THA
   gene expression data.
Acknowledgements