Background: Pleural dissemination of pseudomyxoma peritonei (PMP) is considered an advanced/terminal disease. We review our experience with thoracic cytoreductive surgery (CRS) and intraoperative hyperthermic intrathoracic chemotherapy (HITHOC) for the treatment of pleural recurrence of PMP following previous intraabdominal surgery. Patients and Methods: An observational study of five patients with pleural dissemination from PMP treated with thoracic CRS and intraoperative HITHOC with mitomycin C for 90 min at 41.5C. Results: There were three men. The mean age was 46.5 (10.5) years. Postoperatively, one patient developed Grade 1, one patient developed Grade III and one patient developed Grade IV postoperative complication. Two patients had an unremarkable postoperative recovery. Only one patient has died 38 months since treatment from abdominal complication of this disease with no evidence of thoracic disease. The four surviving patients are still alive 4.6-47.4 months after treatment. Two patients have evidence of an intraabdominal recurrence. Conclusions: Thoracic CRS and intraoperative HITHOC is a safe and effective procedure to treat pleural dissemination from PMP. Long-term disease-free survival can be achieved from this treatment for which no other potentially curative therapy has been described.