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• W2600188037 abstract "10060 Background: Pleuropulmonary blastoma (PPB) is a rare malignancy of the lung presenting in young children. The International PPB Registry (IPBBR) has pathologically confirmed more than 400 cases of PPB. Three pathologic subtypes correlate with age and outcome: Type I, a purely cystic lesion often cured by surgery alone; Type II, a combined cystic and solid lesion; and Type III, a purely solid high-grade sarcoma. Previously reported five-year survival rates for Types II and III PPB are 67% and 51%, respectively. Historically, treatment for Types II and III PPB has not been uniform. The IPPBR conducted the first prospective treatment study to evaluate the response of Type II and III PPB to a standardized chemotherapy regimen. Methods: Treatment records were obtained forpatients with Type II and III PPB who enrolled on the IPBBR Treatment and Biology registry. A regimen of 36 weeks of ifosfamide, vincristine, actinomycin and doxorubicin (IVADo) was recommended. Sixty-two patients were treated on the IVADo regimen; 42 completed 3 or more cycles of induction IVADo and were considered fully assessable for analyses. The IVADo group was compared as a group to 200 historical IPPBR confirmed PPB patients who received regimens other than IVADo for both event-free survival (EFS) and overall survival (OS). Results: Of the 42 patients, 25 (60%) were males and 17 (40%) females; median age at diagnosis was 35 months (range 19-83). There were 24 (57%) Type II; 4 (10%) Type II/III, and 14 (33%) Type III. At a median follow-up of 28 months (range 2-71), 10 (24%) relapsed; 6 (14%) died at 12-24 months. Compared to the historical group, the IVADo group had better EFS (Log-rank test, p=0.008) and better OS (p=0.04). At 5 years, IVADo had better EFS: 70.8% (95% CI 55.3-86.3%) vs. 44.7% (95% CI 37.1-52.4%), and better OS 79.2% (64.1-94.2%) vs. 60.1% (52.3-67.9%). Conclusions: To date, the IPPBR treatment study represents the only cohort of patients with Type II and Type III PPB that have been treated uniformly. The standardized use of IVADo showed improvement in EFS and OS compared to the historical group. Importantly, the follow-up of the IVADo cohort is shorter, and there are late events and deaths in the historical group, limiting our conclusion. Clinical trial information: NCT01464606." @default.
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• W2600188037 date "2014-05-20" @default.
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• W2600188037 title "IVADo treatment of type II and type III pleuropulmonary blastoma (PPB): A report from the International PPB Registry." @default.
• W2600188037 doi "https://doi.org/10.1200/jco.2014.32.15_suppl.10060" @default.
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