The function and regulation of heat shock transcription factor in Cryptococcus

✅ 全文

隐球菌中热休克转录因子的功能与调控

作者 Chenhao Suo; Yiru Gao; Chen Ding; Tianshu Sun 期刊 Frontiers in Cellular and Infection Microbiology 发表日期 2023 ISSN 2235-2988 DOI 10.3389/fcimb.2023.1195968 类型 原创研究 (Original Research)

📄 英文摘要 English Abstract

EN

Cryptococcus species are opportunistic human fungal pathogens. Survival in a hostile environment, such as the elevated body temperatures of transmitting animals and humans, is crucial for Cryptococcus infection. Numerous intriguing investigations have shown that the Hsf family of thermotolerance transcription regulators plays a crucial role in the pathogen-host axis of Cryptococcus . Although Hsf1 is known to be a master regulator of the heat shock response through the activation of gene expression of heat shock proteins (Hsps). Hsf1 and other Hsfs are multifaceted transcription regulators that regulate the expression of genes involved in protein chaperones, metabolism, cell signal transduction, and the electron transfer chain. In Saccharomyces cerevisiae , a model organism, Hsf1’s working mechanism has been intensively examined. Nonetheless, the link between Hsfs and Cryptococcus pathogenicity remains poorly understood. This review will focus on the transcriptional regulation of Hsf function in Cryptococcus , as well as potential antifungal treatments targeting Hsf proteins.

📄 中文摘要 Chinese Abstract

中文
隐球菌属是一类机会性人类真菌病原体。在恶劣环境中生存,例如在传播动物和人类体内较高的体温条件下存活,对于隐球菌感染至关重要。大量引人注目的研究表明,耐热性转录调控因子Hsf家族在隐球菌的病原体-宿主轴中发挥着关键作用。尽管已知Hsf1是通过激活热激蛋白(Hsps)基因表达来调控热激反应的主调控因子,但Hsf1及其他Hsfs是多方面的转录调控因子,可调控涉及蛋白质伴侣、代谢、细胞信号转导和电子传递链的基因表达。在酿酒酵母(_Saccharomyces cerevisiae_)中,Hsf1的工作机制已被深入研究,但Hsfs与隐球菌致病性之间的联系仍知之甚少。新发和再发真菌病原体是人类和动物疾病及死亡的主要原因之一。约15%的艾滋病相关死亡由隐球菌感染引起,每年导致约220,000人死亡。抗真菌药物选择有限,且耐药性的快速发展常常阻碍真菌感染的治疗。

📋 英文结构化总结 English Structured Summary

全文整理

EN

Header:

Background

_Cryptococcus_ species are opportunistic human fungal pathogens. Survival in a hostile environment, such as the elevated body temperatures of transmitting animals and humans, is crucial for _Cryptococcus_ infection. Numerous intriguing investigations have shown that the Hsf family of thermotolerance transcription regulators plays a crucial role in the pathogen-host axis of _Cryptococcus_. Although Hsf1 is known to be a master regulator of the heat shock response through the activation of gene expression of heat shock proteins (Hsps), Hsf1 and other Hsfs are multifaceted transcription regulators that regulate the expression of genes involved in protein chaperones, metabolism, cell signal transduction, and the electron transfer chain. In _Saccharomyces cerevisiae_, Hsf1’s working mechanism has been intensively examined, but the link between Hsfs and _Cryptococcus_ pathogenicity remains poorly understood. Emerging and re-emerging fungal pathogens are one of the leading causes of human and animal illness and mortality. Around 15% of AIDS-related deaths are caused by _Cryptococcus_ infections, resulting in approximately 220,000 deaths per year. Antifungal medication options are limited, and the rapid development of drug resistance frequently impedes fungal treatment.

Header:

Methods

N/A - Review article

Header:

Results

Heat shock transcription factors (Hsfs) are a family of highly conserved DNA-binding proteins that provide thermoprotection to cells by activating the expression of canonical target genes encoding heat shock proteins. The activation of Hsf1 is dependent on an increase in temperature, which dissociates the Hsf1 inhibitory complex in the cytosol compartment to produce the DNA-binding-competent homotrimer complex. Hsf1 complex binds heat shock element sequences of its target genes, then activates downstream gene expression in response to intracellular stressors such as protein misfolding and oxidative damage. The Hsf family and its regulatory mechanism are highly conserved between _Saccharomyces cerevisiae_ (which contains one _HSF_ gene) and human cells (which contain six _HSF_ genes). _C. neoformans_ _HSF1_, similar to that of _S. cerevisiae_, is an essential gene for cell growth under normal conditions. Yang et al. observed an unanticipated down-regulation of _HSF1_ gene expression in response to elevated temperature.

Header:

Data Summary

Around 15% of AIDS-related deaths are caused by _Cryptococcus_ infections, resulting in approximately 220,000 deaths per year. Pigeon body temperatures are 42 ± 1.3°C. _Saccharomyces cerevisiae_ contains one _HSF_ gene, while human cells contain six _HSF_ genes (_HSF1_, _HSF2_, _HSF4_, _HSF5_, _HSFX_ and _HSFY_). The _HSF1_ endogenous promoter in _C. neoformans_ cannot be replaced with an inducible promoter without inhibiting cell development.

Header:

Conclusions

The Hsf family and its regulatory mechanism are highly conserved between _Saccharomyces cerevisiae_ and human cells. Hsf1 is a crucial element in the development of human diseases, although little is known about its regulation in the production of fungal virulence factors. This review focuses on the function of the Hsf family in _Cryptococcus_ and elucidates its function and control in _Cryptococcus_ pathogenicity. The link between Hsfs and _Cryptococcus_ pathogenicity remains poorly understood, underscoring the need for further investigation.

Header:

Practical Significance

This review discusses potential antifungal treatments targeting Hsf proteins. The ability of _Cryptococcus_ to survive at human body temperature of 37°C as well as in even harsher temperature conditions is a key factor contributing to its success as a fungal pathogen, and understanding the Hsf regulation axis may inform new therapeutic strategies.

📋 中文结构化总结 Chinese Structured Summary

中文

背景:

隐球菌属是一类机会性人类真菌病原体。在恶劣环境中生存,例如在传播动物和人类体内较高的体温条件下存活,对于隐球菌感染至关重要。大量引人注目的研究表明,耐热性转录调控因子Hsf家族在隐球菌的病原体-宿主轴中发挥着关键作用。尽管已知Hsf1是通过激活热激蛋白(Hsps)基因表达来调控热激反应的主调控因子,但Hsf1及其他Hsfs是多方面的转录调控因子,可调控涉及蛋白质伴侣、代谢、细胞信号转导和电子传递链的基因表达。在酿酒酵母(_Saccharomyces cerevisiae_)中,Hsf1的工作机制已被深入研究,但Hsfs与隐球菌致病性之间的联系仍知之甚少。新发和再发真菌病原体是人类和动物疾病及死亡的主要原因之一。约15%的艾滋病相关死亡由隐球菌感染引起,每年导致约220,000人死亡。抗真菌药物选择有限,且耐药性的快速发展常常阻碍真菌感染的治疗。

方法:

不适用——综述类文章

结果:

热激转录因子(Hsfs)是一类高度保守的DNA结合蛋白家族,通过激活编码热激蛋白的经典靶基因的表达来为细胞提供热保护。Hsf1的激活依赖于温度升高,温度升高会使细胞质区室中的Hsf1抑制复合物解离,从而产生具有DNA结合能力的同源三聚体复合物。Hsf1复合物与其靶基因的热激元件序列结合,然后在响应细胞内胁迫因子(如蛋白质错误折叠和氧化损伤)时激活下游基因的表达。Hsf家族及其调控机制在酿酒酵母(含一个_HSF_基因)和人类细胞(含六个_HSF_基因)之间高度保守。新型隐球菌(_C. neoformans_)的_HSF1_与酿酒酵母类似,是正常条件下细胞生长所必需的基因。Yang等人观察到,在温度升高时_HSF1_基因表达出现了意料之外的下调。

数据摘要:

约15%的艾滋病相关死亡由隐球菌感染引起,每年导致约220,000人死亡。鸽子体温为42 ± 1.3°C。酿酒酵母含一个_HSF_基因,而人类细胞含六个_HSF_基因(_HSF1_、_HSF2_、_HSF4_、_HSF5_、_HSFX_和_HSFY_)。新型隐球菌的_HSF1_内源性启动子若被诱导型启动子替换,会抑制细胞发育。

结论:

Hsf家族及其调控机制在酿酒酵母与人类细胞之间高度保守。Hsf1是人类疾病发生发展中的关键要素,但对其在真菌毒力因子产生中的调控作用知之甚少。本综述聚焦于Hsf家族在隐球菌中的功能,并阐明其在隐球菌致病性中的功能与调控机制。Hsfs与隐球菌致病性之间的联系仍不清楚,凸显了进一步研究的必要性。

实际意义:

本综述讨论了靶向Hsf蛋白的潜在抗真菌治疗策略。隐球菌能够在人体体温37°C乃至更严苛的温度条件下存活,是其作为真菌病原体成功致病的关键因素,而理解Hsf调控轴可能为新的治疗策略提供理论依据。

📖 英文全文 English Full Text

EN

TYPE Mini Review PUBLISHED 24 April 2023 DOI 10.3389/fcimb.2023.1195968 OPEN ACCESS EDITED BY Tong-Bao Liu, Southwest University, China REVIEWED BY Ning-Ning Liu, Shanghai Jiao Tong University, China *CORRESPONDENCE

Tianshu Sun sun_tianshu@163.com Chen Ding dingchen@mail.neu.edu.cn

The function and regulation of heat shock transcription factor in Cryptococcus Chenhao Suo 1, Yiru Gao 1, Chen Ding 1* and Tianshu Sun 2,3* 1 College of Life and Health Sciences, Northeastern University, Shenyang, China, 2 Medical Research Center, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Science, Beijing, China, 3 Beijing Key Laboratory for Mechanisms Research and Precision Diagnosis of Invasive Fungal Diseases, Beijing, China

This article was submitted to Fungal Pathogenesis, a section of the journal Frontiers in Cellular and Infection Microbiology RECEIVED 29 March 2023 ACCEPTED 10 April 2023 PUBLISHED 24 April 2023 CITATION

Suo C, Gao Y, Ding C and Sun T (2023) The function and regulation of heat shock transcription factor in Cryptococcus. Front. Cell. Infect. Microbiol. 13:1195968. doi: 10.3389/fcimb.2023.1195968 COPYRIGHT

© 2023 Suo, Gao, Ding and Sun. This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.

Cryptococcus species are opportunistic human fungal pathogens. Survival in a hostile environment, such as the elevated body temperatures of transmitting animals and humans, is crucial for Cryptococcus infection. Numerous intriguing investigations have shown that the Hsf family of thermotolerance transcription regulators plays a crucial role in the pathogen-host axis of Cryptococcus. Although Hsf1 is known to be a master regulator of the heat shock response through the activation of gene expression of heat shock proteins (Hsps). Hsf1 and other Hsfs are multifaceted transcription regulators that regulate the expression of genes involved in protein chaperones, metabolism, cell signal transduction, and the electron transfer chain. In Saccharomyces cerevisiae, a model organism, Hsf1’s working mechanism has been intensively examined. Nonetheless, the link between Hsfs and Cryptococcus pathogenicity remains poorly understood. This review will focus on the transcriptional regulation of Hsf function in Cryptococcus, as well as potential antifungal treatments targeting Hsf proteins. KEYWORDS

heat shock factor, protein chaperone, Cryptococcus, fungal infection, thermotolerance

Introduction Emerging and re-emerging fungal pathogens are one of the leading causes of human and animal illness and mortality (Akerfelt et al., 2010; Anckar and Sistonen, 2011; Brown et al., 2012; Fisher et al., 2012; Vos et al., 2012; Denning and Bromley, 2015; Gomez-Pastor et al., 2018). Cryptococcus species are encapsulated opportunistic pathogenic fungi that threaten human societies. Around 15% of AIDS-related deaths are caused by Cryptococcus infections, resulting in approximately 220,000 deaths per year (Idnurm et al., 2005; Kronstad et al., 2012; Erwig and Gow, 2016; Rajasingham et al., 2017). Recent researches have shown that infections caused by Cryptococcus species are a substantial public health concern in the Pacific Northwest of Northern America, Europe, Africa, and China (Byrnes et al., 2010; Byrnes et al., 2011; May et al., 2016; Liu et al., 2017). Nevertheless, antifungal medication options are limited, and the rapid development of drug resistance frequently impedes fungal treatment (Coste et al., 2004; Silver et al., 2004; Morschhauser et al., 2007; Frontiers in Cellular and Infection Microbiology

shock element sequences of its target genes, which then activates downstream gene expression in response to intracellular stressors such as protein misfolding and oxidative damage. We now know that the Hsf family and its regulatory mechanism are highly conserved between Saccharomyces cerevisiae (which contains one HSF gene) and human cells, which contain six HSF genes (HSF1, HSF2, HSF4, HSF5, HSFX and HSFY) (Gomez-Pastor et al., 2018). Hsf1 is the most extensively studied of these Hsf transcription factors. Studies have indicated that Hsf1 is a crucial element in the development of human diseases, although little is known about its regulation in the production of fungal virulence factors (Neef et al., 2010; Neef et al., 2011; Neef et al., 2014; Gomez-Pastor et al., 2018; Dong et al., 2019; Dong et al., 2020). In mammals, for instance, the inhibition or elevation of Hsf1 results in the development of neurological disorders and cancer, respectively. C. neoformans HSF1, similar to that of S. cerevisiae, is an essential gene for cell growth under normal conditions, as the HSF1 endogenous promoter cannot be replaced with an inducible promoter (a galactose inducible promoter) without inhibiting cell development (Yang et al., 2017; Gao et al., 2022). Yang et al. studied the regulation mechanism of C. neoformans HSF1 for the first time in a thermotolerance transcriptome analysis using DNA microarrays. They observed an unanticipated down-regulation of HSF1 gene expression in response to elevated temperature. Another investigation of HSF1 further supported this observation in C. neoformans (Gao et al., 2022). HSP90 gene expression is strongly induced by temperature shifts despite the fact that HSP90 has a conventional Hsf1 binding motif and is a direct target of Hsf1. Curiously, HSF1 overexpression conferred tolerance to deadly temperature conditions (Yang et al., 2017). Why does C. neoformans inhibit the expression of the HSF1 gene if it is essential for thermotolerance? In one scenario, the dramatic increase of Hsp90 inhibits C. neoformans HSF1 gene expression in order to prevent its autoregulation of transcription (GomezPastor et al., 2017). However, Hsf1 chromatin immunoprecipitation PCR analysis revealed no DNA amplification of its own promoter sequence, indicating that C. neoformans HSF1 is regulated differently than in other fungal species (Yang et al., 2017). In addition to regulating protein chaperones, a DNA microarray study revealed that C. neoformans HSF1 acts as both a

Dunkel et al., 2008; van der Linden et al., 2011; Kwon-Chung and Chang, 2012; Billmyre et al., 2020; Li et al., 2020; Priest et al., 2022). Infections caused by Cryptococcus begin in the lung tissue and subsequently spread to the central nervous system, resulting in fatal meningitis (Hull and Heitman, 2002; Kronstad et al., 2011; Kronstad et al., 2013). Machineries, such as those involved in morphological alterations, capsule formation, nutrition acquisition, and thermotolerance, play an essential role in regulating the colonization, invasion, and replication of cells in host tissues (Idnurm et al., 2005; Lee et al., 2013; Gerwien et al., 2018; Verbancic et al., 2018; Gao et al., 2022). The ability of Cryptococcus to survive at human body temperature of 37 °C as well as in even harsher temperature conditions, such as those found in the transmission animal species, such as pigeons, whose body temperatures are 42 ± 1.3°C (Adams et al., 1999), is a key factor that contributes to its success as a fungal pathogen (Figure 1). The heat shock transcription factor (Hsf) regulation axis is one of the most extensively studied systems controlling thermotolerance in eukaryotic cells, involving both canonical and non-canonical transcription regulation patterns (Akerfelt et al., 2010; Anckar and Sistonen, 2011; Mendillo et al., 2012; Gomez-Pastor et al., 2018; Veri et al., 2018). Numerous outstanding reviews provide a comprehensive overview of the relationship between Hsf activity and fungal biology (Yamamoto et al., 2007; Morano et al., 2012; Veri et al., 2018). Hence, this review focuses on the function of the Hsf family in Cryptococcus and elucidates its function and control in Cryptococcus pathogenicity.

Canonical function of heat shock factor in Cryptococcus Heat shock transcription factors (Hsfs) are a family of highly conserved DNA-binding proteins that provide thermoprotection to cells by activating the expression of canonical target genes encoding heat shock proteins (Hsps) (Akerfelt et al., 2010; Anckar and Sistonen, 2011). The activation of Hsf1 is dependent on an increase in temperature, which dissociates the Hsf1 inhibitory complex in the cytosol compartment to produce the DNAbinding-competent homotrimer complex (Akerfelt et al., 2010; Anckar and Sistonen, 2011) (Figure 2). Hsf1 complex binds heat

Cryptococcus infection model. Both humans and pigeons are hosts for Cryptococcus species, which specutilize thermotolerance to survive in pigeons with a body temperature of 42 ± 1.3°C and in humans with a temperature range of 36 to 42°C (Cramer et al., 2022). This figure was created with BioRender.com.

Frontiers in Cellular and Infection Microbiology 02 frontiersin.org Suo et al. 10.3389/fcimb.2023.1195968 FIGURE 2

Hsf transcription regulation of C. neoformans. Hsf1 and Hsf3 participate in transcriptional regulation of gene expression, however the role of Hsf2 in C. neoformans is unknown. While Hsf1 and Hsf3 are transcription factors confined to the nucleus, Hsf3 is also translocated to the mitochondrion. As investigated in other organisms, Hsf1 in C. neoformans is likely repressed by Hsp90 under normal conditions and then translocated to the nucleus, where it is phosphorylated. The activated Hsf1 is subsequently trimerized to bind to gene promoter regions. Hsf1 of C. neoformans regulates the gene expression of canonical and noncanonical genes involved in glucose metabolism, cell communication, oxidative stress, and signal transduction. It is unknown if C. neoformans Hsf3 trimerizes in response to stimulation or if an Hsf1-Hsf3 complex is produced. Hsf3 in the nucleus of C. neoformans regulates the expression of genes involved in the TCA cycle, the electron transport chain, and the ribosome. ROS levels regulate the activity of mitochondrial Hsf3, which binds to the mitochondrial genome to promote the production of genes encoding the electron transport chain. This figure was created with BioRender.com.

identified, it has been demonstrated that a protein kinase, Sch9, regulates HSF1 gene expression and protein level in a sophisticated way; that is, under basal conditions Sch9 regulates Hsf1 protein levels, whereas under heat shock conditions Sch9 represses HSF1 gene expression. However, the PTM analysis of C. neoformans Hsf1 is greatly hindered by the lack of a thorough study of Hsf1 PTM utilizing pan-antibody coupled mass spectrometry analysis and the inadequacy of the production of specialized antibodies for Hsf1 PTM sites. Recently generated phosphatase and kinase knockout libraries provide key tools to facilitate and accelerate the identification of upstream PTM enzymes of HSF1 and to map the regulation network of this essential transcription factor in C. neoformans (Lee et al., 2016; Jin et al., 2020).

positive and negative transcriptional regulator in response to oxidative stress (Yang et al., 2017). In fact, a high-throughput ChIP sequencing investigation revealed that Hsf1 is a wide transcription regulator capable of DNA binding to noncanonical target genes involved in various biological processes, including glycolysis, cell communication, and signal transduction (Gao et al., 2022). The question of whether Hsf1 is a crucial virulence determinant remains unanswered, as the first experiment with an HSF1 overexpression strain exhibited the same pathogenicity as the wild-type strain. However, given that HSF1 is an essential gene, suppression of C. neoformans HSF1 is likely a promising avenue for the development of anti-cryptococcal treatment. In other organisms, particularly mammals, posttranslational modifications (PTM) such as phosphorylation, acetylation, and SUMOylation have been demonstrated to regulate Hsf1 activity (Westerheide et al., 2009; Asano et al., 2016; Hendriks et al., 2017). Fungal Hsf1 has been found to be phosphorylated (Hoj and Jakobsen, 1994; Liu and Thiele, 1996; Hashikawa and Sakurai, 2004), although additional PTM are still unknown (Figure 2). Hsf1 may not be acetylated in C. neoformans, according to a recent comprehensive acetylome investigation, which failed to detect Hsf1 acetylation (Li et al., 2019). In S. cerevisiae, the transition from a basal level of Hsf1 phosphorylation to a hyperphosphorylation process was assumed to constitute a regulation of Hsf1 transcriptional activity levels. When C. neoformans was exposed to a higher temperature, temporary phosphorylation of Hsf1 was also detected, likely indicating the activation of Hsf1 activity (Yang et al., 2017). Although direct phosphorylation enzymes to C. neoformans Hsf1 have not yet been

Noncanonical function of heat shock transcription factor in Cryptococcus Despite the fact that Hsf1 is the master transcription regulator of canonical HSP gene expression in response to temperature elevation, Hsf1 also regulates a large number of noncanonical target genes (Mendillo et al., 2012; Gao et al., 2022). For example, in mammals, Hsf1 directly coordinates in regulation of gene expression of malignancy, including cell cycle regulation, signaling, metabolism and translation. In addition, other mammalian Hsfs, including human Hsf2, have been shown to coordinate with Hsf1 to form heterotrimer complexes that regulate the expression of noncanonical target genes involved in

Results demonstrated that Hsf3 serves as a thermoprotector of C. neoformans via the regulation of gene expression of key components involved in electron transport chain, particularly the NDUFA5 subunit from complex I and Qcr9 subunit from complex III. Dampening Hsf3 protein level results in downregulation of ETC activity which promotes accumulation of intramitochondrial reactive oxygen species. The heat sensitive growth of the HSF3 deletion C. neoformans strain is readily rescued by overexpression mitochondrial specific superoxide dismutase (SOD2). Furthermore, C. neoformans Hsf3 acts as a redox sensing protein via the oxidation of the 130th cysteine residues. The oxidation by reactive oxygen species improves C. neoformans Hsf3’s mitochondrial DNA binding affinity. In a separate study, mammalian Hsf2 was suggested to be the redox sensor that determines cell fate via its regulation axis of HSF2BTG2-SOD2, in which ROS activation of mammalian Hsf2 triggers the gene expression of BTG2 and SOD2 (Kanugovi Vijayavittal et al., 2022). The major disparity between two sensing mechanisms is that C. neoformans Hsf3 does not control gene expression of SOD2 (Gao et al., 2022). In addition, how does mammalian Hsf2 detect exclusive ROS? It is interesting to determine whether the cysteine residue at position 130 is conserved between two Hsfs.

tumor progression and neurodevelopment (Shinkawa et al., 2011; Mendillo et al., 2012; El Fatimy et al., 2014; Jaeger et al., 2016). According to the traditional paradigm of fungal Hsfs, fungal species contain only one copy of Hsf protein (Gomez-Pastor et al., 2018; Veri et al., 2018; Gao et al., 2022), particularly in the model yeast S. cerevisiae and C. albicans. S. cerevisiae may have deceived our knowledge of the evolution and transcriptional regulation of the Hsf family in fungi. However, a recent study identified at least two Hsf orthologs in the genomes of substantially all fungal species, although their roles in transcription regulation are unknown. Except for S. cerevisiae and C. albicans, all fungal species contain multiple copies of HSF genes. Protein sequence comparisons revealed that the preponderance of fungal pathogen Hsf1s have a high degree of similarity, whereas C. neoformans Hsf3 and Hsfs from other fungal species have a moderate degree of divergence. (Gao et al., 2022). In C. neoformans, expression of all three Hsfs, namely Hsf1, Hsf2 and Hsf3, are all responsive to temperature shift, with reciprocal pattern in transcription regulation; Hsf1 is downregulated under temperature elevation conditions, as Hsf2 and Hsf3 are induced in expression upon heat treatment. Loss of C. neoformans HSF3 decreases the rate of cell survival under lethal temperature conditions and reduces fungal virulence and fitness in a mouse infection model (Gao et al., 2022). C. neoformans Hsf3 does not regulate gene expression of HSPs, instead binds directly to promoter regions of genes involved in carbohydrate metabolism (primarily genes involved in the tricarboxylic acid cycle, TCA) (Figure 2), and this resembles the noncanonical Hsf in mammals. However, C. neoformans Hsf1 and Hsf3 show high similarity in DNA-binding domain and share substantial overlap in target genes (mainly carbohydrate metabolic genes). Since it is now known that mammalian Hsf1 cooperates with other Hsfs, such as human Hsf2 (Jaeger et al., 2016; Gomez-Pastor et al., 2018), it is intriguing to examine whether C. neoformans Hsfs are regulated in a similar manner. Recent examination of mammalian Hsf2 revealed a transcription regulation in aerobic glycolysis (glycolysis) by interacting with euchromatic histone lysine methyltransferase 2 (EHMT2) to inhibit gene expression of fructose-bisphosphatase 1 (Fbp1) (Yang et al., 2019). Remarkably, C. neoformans promoter region of Fbp1 is concurrently bound by both C. neoformans Hsf1 and Hsf3. Five TCA intermediates, including citrate, isocitric acid, malate, fumarate, and alpha-ketoglutarate, are significantly induced when C. neoformans HSF3 is absent. Despite the fact that Hsfs are thought to be nucleus-localized transcription factors, C. neoformans Hsf3 was unexpectedly found in the fungal mitochondrial organelles, showing ubiquitous binding of Hsf3 to the mitochondrial DNA (Gao et al., 2022) (Figure 2). The induction of HSF3 gene expression by mitochondrial stresses, such as heat and inhibitors of the mitochondrial complex, results in ROS overloads in C. neoformans mitochondria. Subsequently, intramitochondrial ROS oxidized Hsf3 to improve its ability to bind to DNA and activate the expression of downstream genes. How Hsf3 is recruited and translocated into the mitochondria of C. neoformans is unknown. It is probably via the universal mitochondrial translocase complex, TOM-TIM (Pfanner and Meijer, 1997). Protein co-IP followed by mass spectrometry identified the physical interaction between C. neoformans Hsf3 and Tim44 protein; however, further analysis is necessary to elucidate the mechanism of Hsf3 translocation.

Hsfs as anti-cryptococcosis targets Cryptococcosis has a significant death rate, yet treatment remains difficult due to the restricted number of antifungal drugs available (van der Linden et al., 2011; Billmyre et al., 2020; Li et al., 2020). In view of the therapeutic limitations, risks, and high costs associated with developing new antifungal medicines, the US Food and Drug Administration (FDA) has classified anti-cryptococcus medications as “orphan drugs,” offering regulatory support by decreasing the requirements for clinical research (Denning and Bromley, 2015). However, anticryptococcal medication resistance develops rapidly, surpassing the development of new therapeutic alternatives. Given that C. neoformans Hsf3 modulates virulence and Hsf1 is critical for regulating fungal growth (Yang et al., 2017), the Hsf family are prospective drug development targets. Given that the protein sequence of C. neoformans Hsf3 is similar to that of human Hsfs (Gao et al., 2022), it is necessary to develop inhibitors that target C. neoformans Hsfs but not human Hsfs (Gao et al., 2022). While the Hsf3 regulation axis may or may not be conserved in Aspergillus and Candida, Hsf1 is the highest priority option for therapeutical targets in the context of a larger understanding of fungal infections. Mammalian researches have offered new light on the production of HSF-targeting compounds for fungi (Neef et al., 2011; Neef et al., 2014; Dong et al., 2020). Mammalian Hsf1 activator and inhibitor have demonstrated efficacy in the treatment of mammalian ailments, such as neurological disorders and malignancies (Neef et al., 2011; Neef et al., 2014; Dong et al., 2020). For instance, a direct Hsf1 inhibitory chemical (Direct Targeted Hsf1 InhiBitor, DTHIB) that physically binds to human Hsf1 and induces its protein degradation suppresses the growth of tumors significantly (Dong et al., 2020). Given the significance of Hsf1 in Cryptococcus, it is intriguing to examine whether DTHIB can bind to and inhibit fungal proliferation in vivo.

📖 中文全文 Chinese Full Text

中文

# 隐球菌中热休克转录因子的功能与调控

**苏辰浩¹,高怡茹¹,丁晨¹*,孙天舒²,³\***

¹ 东北大学生命与健康科学学院,沈阳,中国 ² 中国医学科学院北京协和医院医学研究中心,疑难及罕见病国家重点实验室,北京,中国 ³ 北京市侵袭性真菌病机制研究及精准诊断重点实验室,北京,中国

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## 摘要

隐球菌属(*Cryptococcus*)是一类机会性人类真菌病原体。在恶劣环境中生存,例如在传播媒介动物和人类体内的高温环境中存活,对于隐球菌感染至关重要。大量引人关注的研究表明,耐热性转录调控因子Hsf家族在隐球菌的病原体-宿主互作轴中发挥着关键作用。尽管已知Hsf1是通过激活热休克蛋白(Hsps)基因表达来调控热休克反应的主调控因子,但Hsf1及其他Hsfs是多方面的转录调控因子,可调控涉及蛋白质伴侣、代谢、细胞信号转导和电子传递链的基因表达。在模式生物酿酒酵母(*Saccharomyces cerevisiae*)中,Hsf1的工作机制已被深入研究。然而,Hsfs与隐球菌致病性之间的关联仍知之甚少。本综述将聚焦于隐球菌中Hsf功能的转录调控,以及靶向Hsf蛋白的潜在抗真菌治疗策略。

**关键词:** 热休克因子,蛋白质伴侣,隐球菌,真菌感染,耐热性

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## 引言

新发和再发真菌病原体是人类和动物发病及死亡的主要原因之一(Akerfelt et al., 2010; Anckar and Sistonen, 2011; Brown et al., 2012; Fisher et al., 2012; Vos et al., 2012; Denning and Bromley, 2015; Gomez-Pastor et al., 2018)。隐球菌属是一类具有荚膜的机会性致病真菌,对人类社会构成威胁。约15%的艾滋病相关死亡由隐球菌感染引起,每年导致约220,000例死亡(Idnurm et al., 2005; Kronstad et al., 2012; Erwig and Gow, 2016; Rajasingham et al., 2017)。近期研究表明,隐球菌属感染在北美太平洋西北地区、欧洲、非洲和中国是一个重大的公共卫生问题(Byrnes et al., 2010; Byrnes et al., 2011; May et al., 2016; Liu et al., 2017)。然而,抗真菌药物治疗选择有限,且耐药性的快速发展常常阻碍真菌治疗(Coste et al., 2004; Silver et al., 2004; Morschhauser et al., 2007; Dunkel et al., 2008; van der Linden et al., 2011; Kwon-Chung and Chang, 2012; Billmyre et al., 2020; Li et al., 2020; Priest et al., 2022)。

隐球菌感染始于肺组织,随后扩散至中枢神经系统,导致致命性脑膜炎(Hull and Heitman, 2002; Kronstad et al., 2011; Kronstad et al., 2013)。形态改变、荚膜形成、营养获取和耐热性等机制在调控隐球菌在宿主组织中的定植、侵袭和复制中发挥重要作用(Idnurm et al., 2005; Lee et al., 2013; Gerwien et al., 2018; Verbancic et al., 2018; Gao et al., 2022)。隐球菌在人体温度37°C以及在更严苛温度条件下(如传播媒介物种鸽子体内42 ± 1.3°C的体温)存活的能力,是其作为真菌病原体成功的关键因素(图1)。热休克转录因子(Hsf)调控轴是真核细胞中研究最为广泛的耐热性调控系统之一,涉及经典和非经典转录调控模式(Akerfelt et al., 2010; Anckar and Sistonen, 2011; Mendillo et al., 2012; Gomez-Pastor et al., 2018; Veri et al., 2018)。众多优秀综述全面概述了Hsf活性与真菌生物学之间的关系(Yamamoto et al., 2007; Morano et al., 2012; Veri et al., 2018)。因此,本综述聚焦于隐球菌中Hsf家族的功能,并阐明其在隐球菌致病性中的功能与调控机制。

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## 隐球菌中热休克因子的经典功能

热休克转录因子(Hsfs)是一类高度保守的DNA结合蛋白家族,通过激活编码热休克蛋白(Hsps)的经典靶基因表达来为细胞提供热保护(Akerfelt et al., 2010; Anckar and Sistonen, 2011)。Hsf1的激活依赖于温度升高,温度升高使细胞质区室中的Hsf1抑制复合物解离,从而产生具有DNA结合能力的同源三聚体复合物(Akerfelt et al., 2010; Anckar and Sistonen, 2011)(图2)。Hsf1复合物结合其靶基因的热休克元件序列,随后响应细胞内应激源(如蛋白质错误折叠和氧化损伤)激活下游基因表达。目前已知,Hsf家族及其调控机制在酿酒酵母(含一个*HSF*基因)和人类细胞(含六个*HSF*基因:*HSF1*、*HSF2*、*HSF4*、*HSF5*、*HSFX*和*HSFY*)之间高度保守(Gomez-Pastor et al., 2018)。

Hsf1是这些Hsf转录因子中研究最为深入的。研究表明,Hsf1是人类疾病发生发展的关键要素,但对其在真菌毒力因子产生中的调控作用知之甚少(Neef et al., 2010; Neef et al., 2011; Neef et al., 2014; Gomez-Pastor et al., 2018; Dong et al., 2019; Dong et al., 2020)。例如,在哺乳动物中,Hsf1的抑制或升高分别导致神经系统疾病和癌症的发生。

与酿酒酵母类似,新型隐球菌(*C. neoformans*)*HSF1*是正常条件下细胞生长的必需基因,因为*HSF1*内源启动子不能被诱导型启动子(半乳糖诱导型启动子)替代而不抑制细胞发育(Yang et al., 2017; Gao et al., 2022)。Yang等人在利用DNA微阵列进行的耐热性转录组分析中首次研究了*C. neoformans HSF1*的调控机制。他们观察到在高温条件下*HSF1*基因表达出现意料之外的下调。另一项关于*HSF1*的研究在*C. neoformans*中进一步证实了这一观察结果(Gao et al., 2022)。*HSP90*基因表达在温度变化时强烈被诱导,尽管*HSP90*具有常规的Hsf1结合基序且是Hsf1的直接靶标。值得注意的是,*HSF1*的过表达赋予了对致死温度条件的耐受性(Yang et al., 2017)。如果*HSF1*对耐热性至关重要,为什么*C. neoformans*会抑制*HSF1*基因的表达?在一种情况下,Hsp90的急剧增加抑制*C. neoformans HSF1*基因表达,以防止其转录的自调控(Gomez-Pastor et al., 2017)。然而,Hsf1染色质免疫沉淀PCR分析未显示其自身启动子序列的DNA扩增,表明*C. neoformans HSF1*的调控方式与其他真菌物种不同(Yang et al., 2017)。

除了调控蛋白质伴侣外,一项DNA微阵列研究表明,*C. neoformans HSF1*在氧化应激条件下充当正性和负性转录调控因子(Yang et al., 2017)。事实上,一项高通量ChIP测序研究揭示,Hsf1是一个广泛的转录调控因子,能够与涉及多种生物过程(包括糖酵解、细胞通讯和信号转导)的非经典靶基因结合(Gao et al., 2022)。Hsf1是否是一个关键的毒力决定因子仍是一个未解之谜,因为使用*HSF1*过表达菌株进行的首次实验显示出与野生型菌株相同的致病性。然而,鉴于*HSF1*是一个必需基因,抑制*C. neoformans HSF1*可能是开发抗隐球菌治疗的有前景的途径。

在其他生物中,特别是哺乳动物,翻译后修饰(PTM),如磷酸化、乙酰化和SUMO化,已被证明调控Hsf1活性(Westerheide et al., 2009; Asano et al., 2016; Hendriks et al., 2017)。真菌Hsf1已被发现存在磷酸化(Hoj and Jakobsen, 1994; Liu and Thiele, 1996; Hashikawa and Sakurai, 2004),尽管其他PTM仍未知(图2)。根据一项近期全面的乙酰化蛋白质组学研究,Hsf1在*C. neoformans*中可能不被乙酰化,该研究未能检测到Hsf1的乙酰化(Li et al., 2019)。在*S. cerevisiae*中,从Hsf1磷酸化的基础水平到超磷酸化过程的转变被认为构成了对Hsf1转录活性水平的调控。当*C. neoformans*暴露于较高温度时,也检测到Hsf1的短暂磷酸化,这可能表明Hsf1活性的激活(Yang et al., 2017)。尽管尚未直接鉴定出*C. neoformans* Hsf1的直接磷酸化酶,但已证明一种蛋白激酶Sch9以复杂的方式调控*HSF1*基因表达和蛋白水平;即在基础条件下Sch9调控Hsf1蛋白水平,而在热休克条件下Sch9抑制*HSF1*基因表达。然而,*C. neoformans* Hsf1的PTM分析受到严重阻碍,原因在于缺乏利用泛抗体结合质谱分析对Hsf1 PTM的全面研究,以及针对Hsf1 PTM位点的特异性抗体生产不足。近期构建的磷酸酶和激酶敲除文库提供了关键工具,有助于加速鉴定*HSF1*的上游PTM酶,并绘制这一必需转录因子在*C. neoformans*中的调控网络(Lee et al., 2016; Jin et al., 2020)。

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## 隐球菌中热休克转录因子的非经典功能

尽管Hsf1是响应温度升高调控经典*HSP*基因表达的主转录调控因子,但Hsf1也调控大量非经典靶基因(Mendillo et al., 2012; Gao et al., 2022)。例如,在哺乳动物中,Hsf1直接协调恶性肿瘤相关基因表达的调控,包括细胞周期调控、信号转导、代谢和翻译。此外,其他哺乳动物Hsfs,包括人Hsf2,已被证明与Hsf1协调形成异源三聚体复合物,调控涉及肿瘤进展和神经发育的非经典靶基因的表达(Shinkawa et al., 2011; Mendillo et al., 2012; El Fatimy et al., 2014; Jaeger et al., 2016)。

根据真菌Hsfs的传统范式,真菌物种仅含有一个Hsf蛋白拷贝(Gomez-Pastor et al., 2018; Veri et al., 2018; Gao et al., 2022),特别是在模式酵母*S. cerevisiae*和白念珠菌(*C. albicans*)中。*S. cerevisiae*可能误导了我们对真菌中Hsf家族进化和转录调控的认识。然而,近期研究在绝大多数真菌物种的基因组中鉴定出至少两个Hsf直系同源物,尽管它们在转录调控中的作用尚不清楚。除*S. cerevisiae*和*C. albicans*外,所有真菌物种均含有多个*HSF*基因拷贝。蛋白质序列比较揭示,大多数真菌病原体Hsf1具有高度相似性,而*C. neoformans* Hsf3与其他真菌物种的Hsfs具有中等程度的差异(Gao et al., 2022)。

在*C. neoformans*中,所有三个Hsfs(即Hsf1、Hsf2和Hsf3)的表达均响应温度变化,呈现相互的转录调控模式;Hsf1在温度升高条件下表达下调,而Hsf2和Hsf3在热处理后表达被诱导。*C. neoformans HSF3*的缺失降低了细胞在致死温度条件下的存活率,并降低了真菌在小鼠感染模型中的毒力和适应度(Gao et al., 2022)。*C. neoformans* Hsf3不调控*HSPs*的基因表达,而是直接结合参与碳水化合物代谢(主要是参与三羧酸循环TCA的基因)基因的启动子区域(图2),这类似于哺乳动物中的非经典Hsf。然而,*C. neoformans* Hsf1和Hsf3在DNA结合域上显示出高度相似性,并在靶基因(主要是碳水化合物代谢基因)上具有大量重叠。鉴于目前已知哺乳动物Hsf1与其他Hsfs(如人Hsf2)协作(Jaeger et al., 2016; Gomez-Pastor et al., 2018),研究*C. neoformans* Hsfs是否以类似方式被调控是一个引人关注的问题。近期对哺乳动物Hsf2的研究揭示了其通过与常染色质组蛋白赖氨酸甲基转移酶2(EHMT2)相互作用抑制果糖-1,6-二磷酸酶1(Fbp1)基因表达来调控有氧糖酵解的转录调控(Yang et al., 2019)。值得注意的是,*C. neoformans* Fbp1的启动子区域同时被*C. neoformans* Hsf1和Hsf3结合。当*C. neoformans HSF3*缺失时,五种TCA中间产物(包括柠檬酸、异柠檬酸、苹果酸、富马酸和α-酮戊二酸)被显著诱导。

尽管Hsfs被认为是定位于细胞核的转录因子,但*C. neoformans* Hsf3意外地在真菌线粒体细胞器中被发现,显示出Hsf3与线粒体DNA的广泛结合(Gao et al., 2022)(图2)。线粒体应激(如热应激和线粒体复合物抑制剂)对*HSF3*基因表达的诱导导致*C. neoformans*线粒体中ROS超载。随后,线粒体内ROS氧化Hsf3,提高其结合DNA并激活下游基因表达的能力。Hsf3如何被招募并转位至*C. neoformans*的线粒体尚不清楚。可能通过通用线粒体转位酶复合物TOM-TIM实现(Pfanner and Meijer, 1997)。蛋白质免疫共沉淀结合质谱分析鉴定出*C. neoformans* Hsf3与Tim44蛋白之间的物理相互作用;然而,需要进一步分析以阐明Hsf3转位的机制。

结果表明,Hsf3通过调控电子传递链关键组分的基因表达作为*C. neoformans*的热保护因子,特别是复合物I的NDUFA5亚基和复合物III的Qcr9亚基。降低Hsf3蛋白水平导致ETC活性下调,促进线粒体内活性氧的积累。过表达线粒体特异性超氧化物歧化酶(SOD2)可轻易挽救*HSF3*缺失*C. neoformans*菌株的热敏感生长。此外,*C. neoformans* Hsf3通过第130位半胱氨酸残基的氧化充当氧化还原传感蛋白。活性氧的氧化提高了*C. neoformans* Hsf3的线粒体DNA结合亲和力。在另一项研究中,哺乳动物Hsf2被建议为通过其HSF2-BTG2-SOD2调控轴决定细胞命运的氧化还原传感器,其中哺乳动物Hsf2的ROS激活触发*BTG2*和*SOD2*的基因表达(Kanugovi Vijayavittal et al., 2022)。两种传感机制之间的主要差异在于*C. neoformans* Hsf3不控制*SOD2*的基因表达(Gao et al., 2022)。此外,哺乳动物Hsf2如何特异性检测ROS?确定第130位半胱氨酸残基在两个Hsfs之间是否保守是一个有趣的问题。

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## Hsfs作为抗隐球菌病的靶点

隐球菌病具有较高的死亡率,但由于可用的抗真菌药物数量有限,治疗仍然困难(van der Linden et al., 2011; Billmyre et al., 2020; Li et al., 2020)。鉴于治疗局限性和开发新抗真菌药物的风险及高昂成本,美国食品药品监督管理局(FDA)已将抗隐球菌药物归类为"孤儿药",通过降低临床研究要求提供监管支持(Denning and Bromley, 2015)。然而,抗隐球菌药物耐药性的产生速度超过了新治疗替代方案的开发速度。鉴于*C. neoformans* Hsf3调控毒力且Hsf1对调控真菌生长至关重要(Yang et al., 2017),Hsf家族是有前景的药物开发靶点。由于*C. neoformans* Hsf3的蛋白质序列与人Hsfs相似(Gao et al., 2022),有必要开发靶向*C. neoformans* Hsfs但不靶向人Hsfs的抑制剂(Gao et al., 2022)。虽然Hsf3调控轴在曲霉属(*Aspergillus*)和念珠菌属(*Candida*)中可能保守也可能不保守,但在更广泛的真菌感染认知背景下,Hsf1是治疗靶点的最高优先选择。哺乳动物研究为开发靶向真菌HSF的化合物提供了新思路(Neef et al., 2011; Neef et al., 2014; Dong et al., 2020)。哺乳动物Hsf1激活剂和抑制剂已在治疗哺乳动物疾病(如神经系统疾病和恶性肿瘤)中显示出疗效(Neef et al., 2011; Neef et al., 2014; Dong et al., 2020)。例如,一种直接Hsf1抑制性化合物(直接靶向Hsf1抑制剂,DTHIB),能与人Hsf1物理结合并诱导其蛋白降解,显著抑制肿瘤生长(Dong et al., 2020)。鉴于Hsf1在隐球菌中的重要性,研究DTHIB是否能在体内结合并抑制真菌增殖是一个引人关注的问题。