BACKGROUND: The gold standard treatment for incisional ventral hernia (IVH) is a surgical repair with mesh employment, nevertheless this procedure is burdened by several post-operative complications; among these latter, giant pseudocyst of abdominal wall (GPAW) formation is one of the most rare and its etiopathogenesis remains unclear. CASE REPORT: We describe the case report of a 36 years old, diabetic and obese woman, previously underwent to a csection and IVH repair with on-lay mesh placement, presented to our unit with an asymptomatic left iliac fossa swelling. At ultrasound (US) and CT scan examination it appears to be a subcutaneous cyst of the anterior abdominal wall associated with a recurrent IVH. Therefore, she underwent to a surgical procedure in order to accomplish a complete excision of the lesion along with a repair of the incisional IVH, with a sub-lay mesh positioning. The extensive dead space resulting from the procedure was managed with a quilting suture. No recurrence or complications have been detected at 2 years follow up. CONCLUSION: GPAWs are a rare clinical entity following IVH repair, which occur commonly in female obese patients treated with on-lay mesh positioning. The only effective and definitive treatment is a complete surgical excision along with a correct management of the dead space resulting from the surgical procedure, in order to reduce the recurrence rate.