Successful treatment of metastatic cerebral recurrence of pleuropulmonary blastoma

Pleuropulmonary blastoma (PPB) is a rare pediatric tumor. The central nervous system (CNS) is the most common site of extrathoracic metastasis. The prognosis of PPB patients with CNS metastases is dismal: most patients die within one year after recurrence. Here, we describe two patients diagnosed wi...

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Published inPediatric blood & cancer Vol. 66; no. 5; pp. e27628 - n/a
Main Authors Nakano, Yoshiko, Gatell, Sandra Pisa, Schultz, Kris Ann P., Carrillo, Thais Murciano, Fujisaki, Hiroyuki, Okada, Keiko, Horiike, Masaki, Nakamura, Tetsuro, Watanabe, Yusuke, Matsusaka, Yasuhiro, Sakamoto, Hiroaki, Fukushima, Hiroko, Inoue, Takeshi, Williams, Gretchen M., Hill, D. Ashley, Hara, Junichi
Format Journal Article
LanguageEnglish
Published United States Wiley Subscription Services, Inc 01.05.2019
Subjects
Brain Neoplasms - secondary
Brain Neoplasms - therapy
Central nervous system
Chemotherapy
Child, Preschool
CNS relapse
Combined Modality Therapy
Female
Hematology
Humans
Metastases
Metastasis
metronomic therapy
Neoplasm Recurrence, Local - pathology
Neoplasm Recurrence, Local - therapy
Oncology
Patients
Pediatrics
pleuropulmonary blastoma
Prognosis
Pulmonary Blastoma - pathology
Pulmonary Blastoma - therapy
Radiation therapy
Remission
treatment
treatment
CNS relapse
pleuropulmonary blastoma
metronomic therapy
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Summary:Pleuropulmonary blastoma (PPB) is a rare pediatric tumor. The central nervous system (CNS) is the most common site of extrathoracic metastasis. The prognosis of PPB patients with CNS metastases is dismal: most patients die within one year after recurrence. Here, we describe two patients diagnosed with PPB who developed intracranial recurrences shortly after the end of their initial treatment and were successfully treated by gross total resection, radiotherapy, and chemotherapy. Both patients are in complete remission four and three years after recurrence. Although an optimal regimen remains to be determined, these cases demonstrate that PPB with CNS metastases is potentially curable.
Bibliography:Funding information
DICER1
Y.N., S.P.G., and K.A.P.S. contributed to this work.
Registry is supported by the Pine Tree Apple Classic Fund, the Pediatric Cancer Research Foundation, Children's Minnesota Foundation and the Rein in Sarcoma Foundation. DAH and KAS are supported in part by the National Institutes of Health grant NCI R01CA143167 and The Parson's Foundation (DAH).
The International PPB/
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ISSN:1545-5009
1545-5017
DOI:10.1002/pbc.27628