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Table 1.

Mutation analysis of LM-derived cell-lines.

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Table 1 Expand

Fig 1.

Culture of lymphatic endothelial cells and fibroblasts.

Immunocytology showing CD31 (green) and PROX1 (magenta). Nuclei are counter-stained with DAPI (blue). A) Foreskin-derived HD-LECc4; B) Foreskin-derived HD-LECc2; C) Patient-derived Ly-LEC-12, D) Patient-derived Ly-LEC-2; E) Patient-derived Ly-LEC-10; F) Patient-derived fibroblasts (Ly-F-12) do not express CD31 or PROX1. Bar = 45 μm in A-D,F, and 90 μm in E.

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Fig 1 Expand

Fig 2.

Patient-derived fibroblasts (Ly-F-12).

A) Phase-contrast image showing typical morphology. B) Anti-αSMA staining. C) Anti-vimentin staining. D) Negative control without primary antibodies. Note that all fibroblasts express αSMA and vimentin. Bar = 50μm.

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Fig 2 Expand

Fig 3.

A,B. Phospho-AKT-Ser473 Western blot analysis of lymphatic endothelial cells.

Note that phospho-AKT-Ser473 (pAKT) is highly expressed in lymphatic malformation-derived LECs (Ly-LEC1, 2, 10, and 12), but not in normal HD-LECc3 and c4. Total AKT (tAKT) is found in all cell lines. Antibodies against α-tubulin were used as loading control.

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Fig 3 Expand

Fig 4.

Phospho-ERK Western blot analysis of lymphatic endothelial cells.

Note that phospho-ERK (pERK) is highly expressed in lymphatic malformation-derived Ly-LECs (Ly-LEC1, 10, and 12), but not in normal HD-LECc3 and c4. Ly-LEC2, which contain a Glu109del in PIK3CA, are similar to normal LECs, which contain clear levels of total ERK (tERK). Antibodies against α-tubulin were used as loading control.

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Fig 4 Expand

Fig 5.

Proliferation studies with PIK3CA inhibitor Buparlisib (BKM120) on Ly-LEC (left side, A—C) and HD-LEC (right side, D—F), after 0, 24, 48 and 72 hours (h).

0 h values were set to 100%. Concentrations are indicated. Shown are the mean values of n = 3 independent experiments with each 5–8 replicates.

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Fig 5 Expand

Fig 6.

Proliferation studies with PIK3CA inhibitor Wortmannin on Ly-LEC (left side, A—C) and HD-LEC (right side, D—F), after 0, 24, 48 and 72 hours (h).

0 h values were set to 100%. Concentrations are indicated. Shown are the mean values of n = 3 independent experiments with each 5–8 replicates.

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Fig 6 Expand

Fig 7.

Proliferation studies with PIK3CA inhibitor Ly294002 on Ly-LEC (left side, A—C) and HD-LEC (right side, D—F), after 0, 24, 48 and 72 hours (h).

0 h values were set to 100%. Concentrations are indicated. Shown are the mean values of n = 3 independent experiments with each 5–8 replicates.

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Fig 7 Expand

Fig 8.

Proliferation studies with PIK3CD inhibitor CAL-101 (Idealisib) on Ly-LEC (left side, A—C) and HD-LEC (right side, D—F), after 0, 24, 48 and 72 hours (h).

0 h values were set to 100%. Concentrations are indicated. Shown are the mean values of n = 3 independent experiments with each 5–8 replicates.

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Fig 8 Expand

Fig 9.

Proliferation studies with AKT inhibitor MK2206 on Ly-LEC (left side, A—C) and HD-LEC (right side, D—F), after 0, 24, 48 and 72 hours (h).

0 h values were set to 100%. Concentrations are indicated. Shown are the mean values of n = 3 independent experiments with each 5–8 replicates.

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Fig 9 Expand

Fig 10.

Proliferation studies with multi-kinase inhibitor Sorafenib on Ly-LEC (left side, A—C) and HD-LEC (right side, D—F), after 0, 24, 48 and 72 hours (h).

0 h values were set to 100%. Concentrations are indicated. Shown are the mean values of n = 3 independent experiments with each 5–8 replicates.

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Fig 10 Expand

Fig 11.

Proliferation studies with mTOR inhibitor rapamycin on Ly-LEC (left side, A—C) and HD-LEC (right side, D—F), after 0, 24, 48 and 72 hours (h).

0 h values were set to 100%. Concentrations are indicated. Shown are the mean values of n = 3 independent experiments with each 5–8 replicates.

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Fig 11 Expand

Table 2.

Primers used for mutation analyses of PIK3CA.

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Table 2 Expand