In our study, the low-dose combination of trametinib (MEK inhibitor) and omipalisib (PI3K/mTOR dual inhibitor) repressed tumor size in vivo for at least five weeks without significant adverse events and may be a durable treatment option for the original patient with MLA and other patients with MLA tumors harboring KRAS and PIK3CA variants. Here, PIK3CA is linked to neoplasm.