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fredag 2 februari 2018

Mitä biomed. kirjaston kirja kertoo BETi.stä?

https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4854653/
Kirjassa, joka kertoo  aktuellista syöpäterapiasta  englanniksi, on artikkeli
Shaokun Shu et Kornelia Polyak, BET Bromodomain Proteins as Cancer Therapeutic Targets.
Kirjoitin siitä muutamia muistiinpanoja ja kuvia. Mutta kun nyt haen netistä löydän myös  asiasta seuraavaa:

PMCID: PMC4854653
NIHMSID: NIHMS742790
Response and resistance to BET bromodomain inhibitors in triple negative breast cancer (TNBC).

(TNBC tarkoittaa ER-, PR- HER2- (Puuttua estrogeenireseptori (ER)ilmentymä, puuttuu progesteronireseptori (PR) ilmentymä ja puuttuu HER2reseptori-ilmentymä (HER2 = Human Epidermal Growth Factor Receptor 2)
Tavallisin BC tyyppi on luminaalinen A tai B ja ne ovat ER+, PR+ (70%) 
Sitten on vielä HER2+, jossa on ERBB2 onkogeeniamplifikaatio). 

Otan nettiartikkelista abstraktin tähän blogiin: 

Triple negative breast cancer (TNBC) is a heterogeneous and clinically aggressive disease for which there is no targeted therapy1-3.
BET bromodomain inhibitors, which have shown efficacy in several models of cancer4-6, have not been evaluated in TNBC. These inhibitors displace BET bromodomain proteins such as BRD4 from chromatin by competing with their acetyllysine  (AcK) recognition modules, leading to inhibition of oncogenic transcriptional programs7-9.
 Here we report the preferential sensitivity of TNBCs to BET bromodomain inhibition in vitro and in vivo, establishing a rationale for clinical investigation and further motivation to understand mechanisms of resistance. In paired cell lines selected for acquired resistance to BET inhibition from previously sensitive TNBCs, we failed to identify gatekeeper mutations, new driver events or drug pump activation. BET-resistant TNBC cells remain dependent on wild-type BRD4, which supports transcription and cell proliferation in a bromodomain-independent manner. Proteomic studies of resistant TNBC identify strong association with MED1 and hyper-phosphorylation of BRD4 attributable to decreased activity of PP2A, identified here as a principal BRD4 serine phosphatase. Together, these studies provide a rationale for BET inhibition in TNBC and present mechanism-based combination strategies to anticipate clinical drug resistance.

To explore non-oncogene addiction to BRD4 in breast cancer, we studied a series of BET bromodomain inhibitors (BBI) across breast cell lines reflecting transcriptionally-defined breast cancer subtypes: luminal, HER2+, and TNBC2,10 as well as MCF10A and MCF12A basal/mesenchymal immortalized mammary epithelial cells (Supplementary Table 1). Potent inhibitory effects were observed preferentially in TNBC lines, compared to more resistant luminal lines (Fig. 1a). Analysis of potency of drug response and subtype or known driver mutations identified the basal subtype as the only significant association (p=0.0475) (Supplementary Table 1 and D.N.S.). BRD4 dependency was confirmed by RNAi and phenocopied BBI (Extended data Fig. 1a-c). JQ1 or BRD4 knock-down induced growth inhibition resulted in G1 arrest and apoptosis (Extended Data Fig. 1d-g). Expression of factors described to mediate JQ1 effect (MYC) or required for TNBC growth (JAK2/STAT3) showed no clear association with JQ1 sensitivity (Extended Data Fig. 1h and Extended Data Fig. 2a,b). JQ1 treatment of TNBC cells induced significant morphologic changes consistent with induction of senescence confirmed by β-galactosidase staining and luminal differentiation evidenced by changes in the expression of basal and luminal markers (Extended Data Fig. 2c-d and Fig. 1b). 

( JQ1 = thienotriazolodiazepine)

Extending the translational significance of these findings, we evaluated the ability of JQ1 to inhibit tumor growth in murine TNBC xenografts. Two week treatment efficiently inhibited established tumor growth from SUM159 and MDA-MB-231 lines, and patient-derived primary human TNBC xenografts (Fig. 1c and Extended Data Fig. 2e,f). Down-regulation of BRD4 using two independent TET-inducible shRNAs produced even more pronounced effects leading to complete tumor regression and failure to regrow even after discontinuing doxycycline treatment (Fig. 1c and Extended Data Fig. 2g). Evidence of BBI-induced basal-to-luminal differentiation was confirmed in vivo (Extended Data Fig. 2f,h).

Using integrated epigenomic analysis (Supplementary Table 2), we identified the direct transcriptional targets of BBI in TNBC. BBI binding was identified at active promoter and enhancer regions using ChemSeq11 for biotinylated JQ1 (Bio-JQ1) enrichment and ChIP-seq for acetyl-histone (H3K27ac) and BRD4 enrichment, with the three marks showing near perfect co-localization (Fig. 1d and Extended Data Fig. 3a). BBI efficiently displaced chromatin-bound BRD4 in treated SUM159 (Fig. 1e and Extended Data Fig. 3b) and in SUM149 cells (Extended Data Fig. 3c). To identify biologically relevant, direct targets of BBI in SUM159 and SUM149 cells, we quantified binding of Bio-JQ1 and BRD4 genome-wide and found strong enrichment at 219 and 159 super-enhancers, respectively (SEs; Fig. 1f and Extended Data Fig. 3d and Supplementary Table 3)8,9,12,13
TFs with known roles in breast cancer, such as POU5F1B/MYC14 and HIF1α15, were evident among top SE-associated genes in both lines. Kinetic effects of JQ1 treatment on gene expression demonstrated preferential SE-associated gene down-regulation (Fig. 1g and Extended Data Fig. 3e,f). Expression changes were observed within 3 hours after JQ1 treatment and, as expected, more genes were significantly down- than up-regulated (Extended Data Fig. 3g-j, and Supplementary Table 4). Unsupervised Metacore16 analysis of JQ1 affected target pathways revealed down-regulation of regulatory and effector genes in anti-apoptotic and JAK/STAT signaling pathways (Extended Data Fig. 3k). These data support selective disruption of SE-associated genes by JQ1, leading to deregulation of coordinated transcriptional pathways involved in cell proliferation, invasion, and survival.
Dissecting resistance to targeted therapy is critical to elucidate mechanisms of drug and target action, and to suggest approaches to treat or anticipate drug resistance in patients. Therefore, we established BBI-resistant TNBC cell lines by long-term culture of both SUM159 and SUM149 cells in escalating JQ1 doses. Low (0.5 μM) and high (2.0 μM) doses of JQ1 severely impaired proliferation of parental SUM159 and SUM149 lines, reducing viable cells after 6 days (Fig. 2a and Extended Data Fig. 3l). In contrast, JQ1-resistant cells (SUM159R and SUM149R) proliferated linearly, even in high JQ1 doses (20 μM) (Fig. 2a and Extended Data Fig. 3l). BBI-resistance is not attributable to drug export, as MDR1 and other transporters are not transcriptionally up-regulated (Extended Fig. 4a), co-incubation with MDR1 inhibitors (verapamil) had no effect (Extended Data Fig. 4b), and structurally divergent BBIs are equally inactive as JQ1 (Fig. 2b). Further support is provided by the equivalent chromatin engagement of BRD4 in sensitive and resistant cells, demonstrated by ChemSeq with Bio-JQ1 (Extended Data Fig. 4c). Notably, BBI-resistant TNBC cells retain sensitivity to compounds from orthogonal active drug classes, such as CXCR2 and JAK2 inhibitors17; establishing specific resistance to BBIs (Extended Data Fig. 4d). Adaptive drug resistance was not attributable to outgrowth of a minor subpopulation of pre-existing resistant cells, as 10 independent single cell-derived clones showed similar resistance profiles to pooled SUM159R cells (Extended Data Fig. 4e). Similar results were obtained in vivo, as SUM159R derived xenografts derived were JQ1 unresponsive (Extended Data Fig. 4f). In all resistant TNBC populations studied, exome sequencing failed to identify alterations in BET bromodomain-encoding genes (e.g., gatekeepers) or known driver genes (parallel pathway activation; Supplementary Table 5).
Absent new genetic alterations, we explored the plausibility of an epigenomic mechanism of resistance. Differential enhancer analysis revealed a significant gain of SEs in resistant SUM159R cells (ChemSeq; Fig. 2c and Supplementary Table 6). The gain of Bio-JQ1 SEs was associated with enrichment for BRD4 binding to these genomic loci (Fig. 2d) and increased transcription of associated genes (Fig. 2e). An upstream/intragenic region of H3k27ac at the BCL-xL locus featured prominently among top gained SEs in SUM159R (Fig. 2f), consistent with increased BCL-xL mRNA and protein expression in resistant cells (Supplementary Table 7, Extended Data Fig. 4g). Functionally, cells with acquired resistance to BBI featured a concordant switch in JQ1 anti-apoptotic response based on dynamic BH3 profiling18,19 (Extended Data Fig. 4h).
Observing emergent enhancers in resistant cells, we assessed whether BBI-resistant TNBC cells retained non-oncogene addiction to BRD4. Notably, we observed loss of SUM159R cell viability upon BRD4 knockdown (Extended Data Fig. 5,b). Together these studies establish persistence of BRD4 addiction despite resistance to bromodomain inhibition, establishing the plausibility of bromodomain-independent recruitment of BRD4 to enhancers in BBI-resistant TNBCs. To test this hypothesis, we performed BRD4 ChIP-seq on sensitive and resistant cells with and without JQ1. JQ1 neither displaced BRD4 from chromatin in SUM159R (Fig. 2g), nor meaningfully influence epigenome structure by H3K27ac ChIP-seq (Extended Data Fig. 5c-g). Notably, several luminal markers (FOXA1, CD24, and luminal cytokeratins) were elevated in SUM159R cells in cell culture and in vivo (Extended Data Fig. 5h,i), supporting a model whereby resistance arises via essential BRD4 recruitment to chromatin in a bromodomain-independent manner. Similar observations were made in SUM149R cells and in TNBC cells inherently resistant to JQ1 (Extended Data Fig. 3h-j; Extended Data Fig. 6a-d), suggesting a general mechanism of epigenomic resistance to BBI.
To disclose potential differences in BRD4-associated complexes between sensitive and resistant SUM159 cells, we performed quantitative proteomics using RIME (rapid immunoprecipitation mass spectrometry of endogenous proteins)20 with and without JQ1. Analysis of BRD4-associated proteins identified relative enrichment of MED1 and BRD3 in JQ1-treated resistant cells (Fig. 3a, Extended Data Fig. 7, and Supplementary Table 8). BRD4 immunoprecipitation followed by immunoblot for MED1 and BRD3 revealed that JQ1 efficiently displaced BRD4 from MED1 in sensitive cells, but not in resistant cells (Fig. 3b), a result confirmed in SUM149 and BBI-resistant SUM149R, as well as inherently resistant TNBC and luminal lines (Extended Data Fig. 8a). Though elevated BRD3 abundance was observed in SUM159R, increased association of BRD4 and BRD3 was not confirmed by immunoblot, (Fig. 3b). To assess functionally whether increased recruitment of BRD4 to chromatin by MED1 underlies resistance to JQ1, we expressed an exogenous bromodomain-inactivated mutant (BDmut) with concomitant knock-down of endogenous BRD4 (Extended Data Fig. 8b,c). Downregulation of endogenous BRD4 decreased cell growth both in parental and resistant cells, which was rescued by enforced expression of wild type BRD4 (Fig. 3c). BDmut BRD4 expression failed to rescue parental SUM159 cells, but supported growth of JQ1-resistant SUM159R consistent with an evident bromodomain-independent mechanism of BRD4 recruitment (Fig. 3c). Next, we assessed the sensitivity of cells expressing BDmut BRD4 to JQ1, observing increased sensitivity to JQ1 In parental SUM159 cells exogenously expressing BDmut (Fig. 3d). In contrast, expression of BDmut BRD4 in SUM159R cells rescued the anti-proliferative effect of JQ1 (Fig. 3e), although this could partially be due to the slower growth of BDmut expressing cells. Together, these studies suggest BBI-resistance is associated with increased binding of BRD4 to MED1, in a bromodomain-independent manner unaffected by JQ1.

Mechanism of BBI-resistance in TNBCs

A recent study reported that the stability and nuclear localization of BRD4 is increased with phosphorylation by casein kinase II (CK2)21. To explore the contribution of BRD4 phosphorylation to BBI-resistance, we performed immunoblot analysis in parental and resistant cells and found a marked increase of phospho-BRD4 (pBRD4) in resistant cells (Fig. 4a and Extended Data Fig. 8d). Small-molecule inhibition of CK2 decreased BRD4 phosphorylation in SUM159 and SUM159R cells (Extended Data Fig. 8e). These results imply BRD4 hyperphosphorylation in resistant cells either due to increased phosphorylation by CK2 or, alternatively, to decreased dephosphorylation by an as yet unidentified BRD4 phosphatase. We therefore first analyzed CK2 activity in parental and resistant cells by performing pan-CK2 substrate immunoblots and detected no significant differences in CK2 activity (Extended Data Fig. 8f).

Regulation and relevance of BRD4 phosphorylation


Inactivation of the PP2A phosphatase tumor suppressor gene occurs commonly in breast cancer and is associated with therapy resistance22; PP2A also often opposes CK2 function23,24. Thus, we investigated whether PP2A may dephosphorylate BRD4 and whether decreased PP2A activity could lead to BBI-resistance. Downregulation of PP2A catalytic subunit (PP2CA) in SUM149 and SUM159 cells led to increased BRD4 phosphorylation, establishing PP2A as a previously unrecognized BRD4 phosphatase (Fig. 4b), further supported by pharmacologic inhibitors of PP2A that showed similar effects (Extended Data Fig. 8g). To strengthen the link between PP2A activity and BBI-resistance, we tested the JQ1 sensitivity of SUM149 cells following the knock-down of PP2A C subunit and determined that downregulation of PP2A decreased JQ1 sensitivity (Fig. 4c). We have collaboratively reported phenothiazine compounds as activators of PP2A enzymatic activity25. Thus, we analyzed pBRD4 levels in SUM159R, SUM149R, and other cell lines after short-term treatment with phenothiazine (PTZ) and detected rapid dephosphorylation of BRD4 (Fig. 4d and Extended Data Fig. 8h). Combined treatment with PTZ and JQ1 overcame BBI-resistance in SUM159R cells (Fig. 4e). To investigate the functional role of BRD4 hyperphosphorylation in BBI-resistance, we analyzed whether BRD4 phosphorylation influences MED1 binding. Indeed, SUM159R cells treated with CK2 inhibitor or PTZ both lead to decreased MED1 abundance in BRD4 immunoprecipitations, suggesting that pBRD4 binds MED1 more efficiently than BRD4 (Fig. 4f,g).
To functionally assess the role for BRD4 phosphorylation in BBI-resistance and MED1 binding, we generated BRD4 constructs encoding mutants that cannot be phosphorylated by CK2 (7 serine to alanine substitutions; “7A mutant”) or mimic constitutive phosphorylation (7 serine to aspartate substitutions; “7D mutant”). We first assessed the ability of these constructs to rescue effects of endogenous BRD4 knock-down in stable cell lines (Extended Data Fig. 8b,c). We observed expression of both 7D and 7A mutants supporting the growth of both parental SUM159 and JQ1-resistant SUM159R cells (Fig. 3d). Next, we analyzed MED1 binding and subcellular localization of 7A and 7D mutants +/− JQ1. We found that the 7A mutant displays weaker MED1 binding compared to WT BRD4 and completely dissociates after JQ1 whereas the 7D mutant seems to have higher affinity for MED1 that unaffected by JQ1 treatment (Fig. 4h and Extended Fig. 8i). Lastly, we assessed the sensitivity of cells expressing 7A or 7D mutant BRD4 to JQ1. In parental SUM159 cells exogenously expressed 7D mutant BRD4 decreased sensitivity to JQ1 whereas the 7A mutant slightly increased sensitivity (Fig. 4i). In contrast, expression of 7A mutant BRD4 in SUM159R cells restored JQ1 sensitivity whereas the 7D mutant showed a modest decrease. These results strongly support the hypothesis that hyperphosphorylation of BRD4 arises from decreased PP2A activity in BBI resistant cells leading to increased binding of BRD4 to MED1, recruitment to chromatin and decreased responsiveness to bromodomain inhibition.
To explore the clinical relevance of phospho-BRD4 (pBRD4) in BET inhibitor-naive TNBC, we performed immunofluorescence analysis of a tissue microarray (TMA) featuring of 89 patient-derived TNBC specimens. First, we validated the pBRD4 immunofluorescence assay by comparing xenografts derived from SUM159 and SUM159R cell lines and detected significantly higher pBRD4 in SUM159R cells (Extended Data Fig. 9a). We detected strong pBRD4 staining among a subset of TNBCs (Extended Data Fig. 9b,c), and variable staining overall that was not correlated with expression of the androgen receptor (AR) and basal cytokeratins (bCK; Extended Fig. 9d,e) and it was not significantly associated with disease outcomes (Supplementary Table 9 and Extended Data Fig. 9f).
To extend the translational relevance of our findings, we conducted synergy studies of JQ1 with molecules targeting BCL-xL (ABT737), a gained super-enhancer in SUM159R cells, and modulators of BRD4 phosphorylation, the CK2 inhibitor CX-4945 and the PP2A activator perphenazine (PPZ). We observed significant synergy between JQ1 and all three compounds studied (Extended Data Fig. 10), establishing a rationale for combination studies of BBI in TNBC to improve response and to anticipate BBI resistance.
BRD4 inhibition has demonstrated efficacy in disparate models of cancer in a rapidly expanding literature. Despite apparent resistance in the vast majority of tumor types, as we observed here in TNBC, mechanisms of BBI-resistance have not been mechanistically explained. As this research was in review, two studies reported moderate emergent resistance to BBI in murine AML associated phenotypically with a stem-like state and WNT pathway activation26,27. Interestingly, in our study TNBCs with more basal/stem cell-like features and WNT pathway activation are more sensitive to BET inhibition, whereas resistant disease emerges as epigenomic adaptation to a more differentiated luminal phenotype. Our findings of persistent BET bromodomain dependency despite BBI-resistance, as well as pBRD4 staining in resistant disease should be studied in these murine AML models and further in human leukemia.
Integrating approaches in epigenomics, proteomics, and chemical biology, we provide an example of epigenomic drug resistance by an epigenetic mechanism, where in BBI resistant cells, decreased PP2A activity leads to hyperphosphorylated BRD4, which binds more strongly to MED1, facilitating a bromodomain-independent chromatin recruitment mechanism. This research proposes putative combination strategies to anticipate and overcome BBI resistance, including pairing with BCL-xL inhibitors (e.g., ABT-737) or CK2 inhibitors, and guides the development of second-generation BBIs that disrupt BET function via orthogonal biophysical or biochemical actions. More immediately, the robust efficacy observed in pre-clinical models supports the development of BET inhibition in TNBC alone, and in combination with mechanism-based targeted therapies.

METHODS

Cell lines and breast tumor tissues

Breast cell lines were obtained from the ATCC and Dr. Steve Ethier, University of Michigan, Ann Arbor, MI (SUM series). Cells were cultured in media recommended by the provider, their identity was confirmed by STR analysis, and they were regularly tested for mycoplasma. Breast tumor samples were collected using protocols approved by the DF/HCC Institutional Review Board, informed consent was obtained from all patients. Tumors were minced with razor blades and digested with stirring for 3-4 hours at 37°C in DMEM/F12 with 2 mg/mL BSA, 2 mg/mL collagenase type IV, and 2 mg/mL hyaluronidase. After digestion, cells were filtered through 500-micron mesh, washed in DMEM/F12 with 5% FBS, frozen in DMEM/F12 with 5% FBS and 10% DMSO, and stored in liquid nitrogen for subsequent xenograft studies. PDX IDC50 was derived from a primary tumor of highly invasive metaplastic TNBC resistant chemo and radiation therapy leading to the rapid death of the patient. Exome sequencing of the tumor and xenograft identified numerous mutations including heterozygous frameshift mutation in PTEN (chr10_89701964-89701964_A) and CDH1 chr16_67400242-67400242_C). PDX EL-12-58 was derived from a liver metastasis of a heavily pretreated basal-like TNBC, Oncopanel mutation testing identified homozygous mutations in BRCA2 (p.S1970*), TP53 (p.I232fs), TSC2, FLT3, and ROS1, and lower frequency mutations in RAD21, JAK3, ARID1B, ARID1A, KDM6A.

High-Throughput Screening of BET Bromodomain Inhibitors in breast cell line panel

We tested a panel of compounds (synthesized in the Bradner lab) in 40 human breast cell lines in a 384-well format at 2,000 cells per well using a semi-automated screen essentially as described5. Cell viability at 72 hr was evaluated using ATPlite (Perkin Elmer).

Synergy Studies

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