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Fig 1.

K27, K63 and M1 chains are involved in Tax-induced IKK activation.

(A) Jurkat cytosolic extracts were incubated with recombinant His6-Tax and ATP (2 mM) in the presence of ubiquitin mutants or HA-ubiquitin (50 μM). The reaction mixtures were analyzed by immunoblotting with the indicated antibodies. (B) Cytosolic extracts were prepared from HEK293T cells expressing a series of dominant-negative mutants of the E2 enzyme and subjected to cell-free analyses. (C) Cytosolic extracts were prepared from WT MEFs or Ubc13fl/fl MEFs expressing Cre and subjected to cell-free analyses with increasing amounts of Tax. The depicted results are representative of three independent experiments.

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Fig 2.

LUBAC is required for Tax-induced IKK activation.

(A-C) Cytosolic extracts were prepared from HOIL-1L-deficient (A), Sharpin-deficient (B), HOIPΔlinear (C) and corresponding WT MEFs and subjected to cell-free analyses. (D) Sharpin-deficient and corresponding WT MEFs were mock-infected or were transduced with retroviruses expressing Tax. The MEFs were treated with MG132 (10 μM) for 1 h, and cell lysates were subjected to immunoblotting with the indicated antibodies. The depicted results are representative of three independent experiments.

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Fig 3.

Tax recruits LUBAC to the IKK complex.

(A) Jurkat cytosolic extracts were incubated with recombinant His6-Tax or His6-M22 in the presence of ATP (2 mM). The reaction mixtures were subjected to immunoprecipitation with an anti-Tax antibody, followed by immunoblotting with the indicated antibodies. (B) Cytosolic extracts prepared from Jurkat cells expressing Flag-tagged NEMO were incubated with recombinant His6-Tax or His6-M22 in the presence of ATP (2 mM). The reaction mixtures were subjected to immunoprecipitation with an anti-Flag antibody, followed by immunoblotting with the indicated antibodies. (C) JPX-9 cells were untreated or treated with CdCl2 (20 μM) for 18 h. Cell lysates were subjected to immunoprecipitation with an anti-Tax antibody, followed by immunoblotting with the indicated antibodies. (D) Jurkat cytosolic extracts were incubated with recombinant His6-Tax and ATP (2 mM) in the presence of ubiquitin mutants or HA-ubiquitin (50 μM). The reaction mixtures were subjected to immunoprecipitation with an anti-Tax antibody, followed by immunoblotting with the indicated antibodies. (E) Recombinant His6-Tax was incubated with glutathione sepharose-bound recombinant GST, GST-HOIL-1L, GST-HOIP or GST-Sharpin. His6-Tax bound to GST-tagged protein was analyzed by immunoblotting with an anti-Tax antibody. Dots denote full-length GST fusion proteins. (F) HEK293T cells were transfected with expression plasmids encoding HOIL-1L-HA or various C-terminal HA-tagged HOIL-1L mutants together with a Tax expression plasmid. After 48 h, cell lysates were prepared and subjected to immunoprecipitation with an anti-HA antibody, followed by immunoblotting with an anti-Tax antibody (upper). A schematic representation of the various mutants of HOIL-1L (lower). (G) HEK293T cells were transfected with expression plasmids encoding Myc-HOIP or various N-terminal Myc-tagged HOIP mutants together with a Tax expression plasmid. After 48 h, cell lysates were prepared and subjected to immunoprecipitation with an anti-Myc antibody, followed by immunoblotting with an anti-Tax antibody (upper). A schematic representation of the various mutants of HOIP (lower). The depicted results are representative of three independent experiments.

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Fig 4.

Tax-induced generation of IKK complex-associated K63/M1-linked hybrid chains is required for IKK activation.

(A) Jurkat cytosolic extracts were incubated with recombinant His6-Tax and ATP (2 mM), and the reaction mixtures were subjected to immunoprecipitation with an anti-NEMO antibody. The immunoprecipitates were left untreated or were treated with recombinant Otubain-1, AMSH, OTULIN or USP2 and analyzed by immunoblotting with anti-Ub (upper) and anti-M1 chain-specific (lower) antibodies. (B) Jurkat cytosolic extracts were incubated with or without recombinant His6-Tax and ATP (2 mM), and the reaction mixtures were subjected to immunoprecipitation with an anti-NEMO antibody. The immunoprecipitates were analyzed via the ubiquitin-AQUA method. The results are given as the mean ± SD (n = 3). (C) Cell-free reactions were performed in the presence of ubiquitin mutants or HA-ubiquitin (50 μM). The reaction mixtures were subjected to immunoprecipitation with an anti-NEMO antibody, followed by immunoblotting with an anti-Ub antibody. (D) Cell-free reactions were performed in the presence of various DUBs. The depicted results are representative of three independent experiments.

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Fig 5.

Tax induces formation of the macromolecular active IKK complex.

(A) Jurkat cytosolic extracts were incubated with recombinant His6-Tax or His6-MEKK1 together with ATP (2 mM) in the presence or absence of IsoT. (B) Cell-free reactions were performed in the presence of WT viral OTU or its catalytic inactive mutant 1A. (C) Jurkat cytosolic extracts were incubated with recombinant His6-Tax or its mutants together with ATP (2 mM). The reaction mixtures were subjected to immunoprecipitation with an anti-Tax antibody, followed by immunoblotting with an anti-Ub antibody. (D) Cytosolic extracts were prepared from NEMO-deficient MEFs reconstituted with human NEMO or its mutant and were subjected to cell-free analyses. (E) Cytosolic extracts were prepared from NEMO-deficient MEFs reconstituted with mouse NEMO or its mutants and were subjected to cell-free analyses. (F) Cell-free reactions were performed, and the reaction mixtures were subjected to Blue native-PAGE, followed by immunoblotting with anti-NEMO (left) and anti-p-IKKα/β (right) antibodies. The depicted results are representative of three independent experiments.

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Fig 6.

LUBAC associates with Tax and is involved in NF-κB activation leading to target gene expression and cell proliferation in HTLV-1-infected cells.

(A) Cell lysates from HUT102 cells were subjected to immunoprecipitation with an anti-Tax antibody or a control antibody, followed by immunoblotting with the indicated antibodies. (B) MT-2 (left) or MT-4 (right) cells were infected with control or Tax shRNA expression lentivirus vector. Cell lysates were subjected to immunoprecipitation with an anti-Tax antibody, followed by immunoblotting with the indicated antibodies. (C) MT-2 (left) or MT-4 (right) cells were transfected with control or HOIP siRNA. Cell lysates were subjected to immunoblotting with an anti-pIKKα/β antibody. (D) MT-4 cells were transfected with control or HOIP siRNA as described in (C). The expression levels of IL6 (left), IL1 (middle) and MMP9 (right) were measured by quantitative real-time RT-PCR. The relative expression was calculated by dividing each expression value by that of a control siRNA treatment. (E) MT-2 (left) or MT-4 (right) cells were infected with HOIP shRNA expression lentivirus vector. The cells were seeded at 1.0×105 cells/well, and cell viability was assessed by trypan blue exclusion assay (upper). The whole cell lysates were subjected to immunoblotting with an anti-HOIP antibody to confirm the knockdown of HOIP (lower). The results shown in (D) and (E) are given as the mean ± SD (n = 3). The depicted results are representative of three independent experiments.

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Fig 7.

A model illustrating HTLV-1 Tax-induced IKK activation.

Upon HTLV-1 infection, the Tax protein is translated from doubly spliced viral mRNA expressed from the provirus. Tax is able to bind to NEMO in the IKK complex and to HOIP and HOIL-1L of LUBAC, thereby forming the LUBAC/Tax/IKK complex, which further binds to the Ubc13/Uev1A E2 complex and unidentified K63 E3 enzymes (X) to generate an inactive “pre-Taxisome”. The K63/M1-linked hybrid polyubiquitin chains generated by LUBAC and X (K63 E3) interact with NEMO through its M1 chain-interacting UBAN domain and K63 chain-interacting NZF domain. This multivalent interaction between NEMO proteins and the hybrid polyubiquitin chains results in the oligomerization of the pre-Taxisome to form the macromolecular Taxisome, which allows close interactions between the IKK complexes, leading to the trans-autophosphorylation-mediated activation of the IKK complex. The K63/M1-linked hybrid polyubiquitin chains may contain recently identified branched chains [65].

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