Cerebral metastasis and other central nervous system complications of pleuropulmonary blastoma

John R. Priest, Jeffrey Magnuson, Gretchen M. Williams, Minnie Abromowitch, Rebecca Byrd, Philippa Sprinz, Marsha Finkelstein, Christopher L. Moertel, D. Ashley Hill

Research output: Contribution to journalArticle

47 Citations (Scopus)

Abstract

Background. Pleuropulmonary blastoma (PPB) is a rare tumor of pleura and lung in young children. Central nervous system (CNS) complications, particularly cerebral parenchymal metastases, occur in aggressive forms of PPB: Types II and III PPB. This article evaluates cerebral and meningeal metastases, cerebrovascular events (CVA) caused by tumor emboli, spinal cord complications, and intracranial second malignancies in PPB. Procedure. International PPB Registry and literature cases were evaluated for CNS events. Cerebral metastasis patients were evaluated for gender, side of origin of PPB, PPB Type, interval from diagnosis to metastasis, status of chest disease, treatment, and outcome. Standard statistical methods were used to calculate the cumulative probability of cerebral metastasis and survival following metastasis. Results. Thirty-nine cases of cerebral metastasis were identified in 5/53 Registry Type II cases, 15/44 Registry Type III cases, and 19/143 literature Type II/III cases. Metastases occurred 1-60, median 11.5 months after diagnosis. Chest disease was controlled in 50% of children at time of metastasis. The cumulative probability of cerebral metastasis by 5 years from diagnosis was 11 % for Type II patients (95%CI (confidence interval): 2-20%) and 54% for Type III patients (95%CI: 31-76%). Seven children survive cerebral metastasis. Other CNS complications were post-operative CVA (five cases), spinal cord invasion or compression (six), leptomeningeal disease (three), and second intracranial malignancies (two). Conclusions. Cerebral metastasis is more frequent in PPB than in other childhood sarcomas. Clinicians should screen for this complication. Diverse other CNS complications are less common and require careful diagnosis.

Original languageEnglish (US)
Pages (from-to)266-273
Number of pages8
JournalPediatric Blood and Cancer
Volume49
Issue number3
DOIs
StatePublished - Sep 1 2007

Fingerprint

Central Nervous System
Neoplasm Metastasis
Registries
Second Primary Neoplasms
Pleuropulmonary blastoma
Thorax
Spinal Cord Neoplasms
Pleura
Embolism
Sarcoma
Spinal Cord
Confidence Intervals
Lung
Survival

Keywords

  • Cerebral metastasis
  • Cerebrovascular accidents
  • Pleuropulmonary blastoma
  • Second intracranial neoplasms
  • Tumor embolism

ASJC Scopus subject areas

  • Pediatrics, Perinatology, and Child Health
  • Hematology
  • Oncology

Cite this

Cerebral metastasis and other central nervous system complications of pleuropulmonary blastoma. / Priest, John R.; Magnuson, Jeffrey; Williams, Gretchen M.; Abromowitch, Minnie; Byrd, Rebecca; Sprinz, Philippa; Finkelstein, Marsha; Moertel, Christopher L.; Hill, D. Ashley.

In: Pediatric Blood and Cancer, Vol. 49, No. 3, 01.09.2007, p. 266-273.

Research output: Contribution to journalArticle

Priest, JR, Magnuson, J, Williams, GM, Abromowitch, M, Byrd, R, Sprinz, P, Finkelstein, M, Moertel, CL & Hill, DA 2007, 'Cerebral metastasis and other central nervous system complications of pleuropulmonary blastoma', Pediatric Blood and Cancer, vol. 49, no. 3, pp. 266-273. https://doi.org/10.1002/pbc.20937
Priest, John R. ; Magnuson, Jeffrey ; Williams, Gretchen M. ; Abromowitch, Minnie ; Byrd, Rebecca ; Sprinz, Philippa ; Finkelstein, Marsha ; Moertel, Christopher L. ; Hill, D. Ashley. / Cerebral metastasis and other central nervous system complications of pleuropulmonary blastoma. In: Pediatric Blood and Cancer. 2007 ; Vol. 49, No. 3. pp. 266-273.
@article{af0da8cd1e7640afb9a4a32a0d460580,
title = "Cerebral metastasis and other central nervous system complications of pleuropulmonary blastoma",
abstract = "Background. Pleuropulmonary blastoma (PPB) is a rare tumor of pleura and lung in young children. Central nervous system (CNS) complications, particularly cerebral parenchymal metastases, occur in aggressive forms of PPB: Types II and III PPB. This article evaluates cerebral and meningeal metastases, cerebrovascular events (CVA) caused by tumor emboli, spinal cord complications, and intracranial second malignancies in PPB. Procedure. International PPB Registry and literature cases were evaluated for CNS events. Cerebral metastasis patients were evaluated for gender, side of origin of PPB, PPB Type, interval from diagnosis to metastasis, status of chest disease, treatment, and outcome. Standard statistical methods were used to calculate the cumulative probability of cerebral metastasis and survival following metastasis. Results. Thirty-nine cases of cerebral metastasis were identified in 5/53 Registry Type II cases, 15/44 Registry Type III cases, and 19/143 literature Type II/III cases. Metastases occurred 1-60, median 11.5 months after diagnosis. Chest disease was controlled in 50{\%} of children at time of metastasis. The cumulative probability of cerebral metastasis by 5 years from diagnosis was 11 {\%} for Type II patients (95{\%}CI (confidence interval): 2-20{\%}) and 54{\%} for Type III patients (95{\%}CI: 31-76{\%}). Seven children survive cerebral metastasis. Other CNS complications were post-operative CVA (five cases), spinal cord invasion or compression (six), leptomeningeal disease (three), and second intracranial malignancies (two). Conclusions. Cerebral metastasis is more frequent in PPB than in other childhood sarcomas. Clinicians should screen for this complication. Diverse other CNS complications are less common and require careful diagnosis.",
keywords = "Cerebral metastasis, Cerebrovascular accidents, Pleuropulmonary blastoma, Second intracranial neoplasms, Tumor embolism",
author = "Priest, {John R.} and Jeffrey Magnuson and Williams, {Gretchen M.} and Minnie Abromowitch and Rebecca Byrd and Philippa Sprinz and Marsha Finkelstein and Moertel, {Christopher L.} and Hill, {D. Ashley}",
year = "2007",
month = "9",
day = "1",
doi = "10.1002/pbc.20937",
language = "English (US)",
volume = "49",
pages = "266--273",
journal = "Pediatric Blood and Cancer",
issn = "1545-5009",
publisher = "Wiley-Liss Inc.",
number = "3",

}

TY - JOUR

T1 - Cerebral metastasis and other central nervous system complications of pleuropulmonary blastoma

AU - Priest, John R.

AU - Magnuson, Jeffrey

AU - Williams, Gretchen M.

AU - Abromowitch, Minnie

AU - Byrd, Rebecca

AU - Sprinz, Philippa

AU - Finkelstein, Marsha

AU - Moertel, Christopher L.

AU - Hill, D. Ashley

PY - 2007/9/1

Y1 - 2007/9/1

N2 - Background. Pleuropulmonary blastoma (PPB) is a rare tumor of pleura and lung in young children. Central nervous system (CNS) complications, particularly cerebral parenchymal metastases, occur in aggressive forms of PPB: Types II and III PPB. This article evaluates cerebral and meningeal metastases, cerebrovascular events (CVA) caused by tumor emboli, spinal cord complications, and intracranial second malignancies in PPB. Procedure. International PPB Registry and literature cases were evaluated for CNS events. Cerebral metastasis patients were evaluated for gender, side of origin of PPB, PPB Type, interval from diagnosis to metastasis, status of chest disease, treatment, and outcome. Standard statistical methods were used to calculate the cumulative probability of cerebral metastasis and survival following metastasis. Results. Thirty-nine cases of cerebral metastasis were identified in 5/53 Registry Type II cases, 15/44 Registry Type III cases, and 19/143 literature Type II/III cases. Metastases occurred 1-60, median 11.5 months after diagnosis. Chest disease was controlled in 50% of children at time of metastasis. The cumulative probability of cerebral metastasis by 5 years from diagnosis was 11 % for Type II patients (95%CI (confidence interval): 2-20%) and 54% for Type III patients (95%CI: 31-76%). Seven children survive cerebral metastasis. Other CNS complications were post-operative CVA (five cases), spinal cord invasion or compression (six), leptomeningeal disease (three), and second intracranial malignancies (two). Conclusions. Cerebral metastasis is more frequent in PPB than in other childhood sarcomas. Clinicians should screen for this complication. Diverse other CNS complications are less common and require careful diagnosis.

AB - Background. Pleuropulmonary blastoma (PPB) is a rare tumor of pleura and lung in young children. Central nervous system (CNS) complications, particularly cerebral parenchymal metastases, occur in aggressive forms of PPB: Types II and III PPB. This article evaluates cerebral and meningeal metastases, cerebrovascular events (CVA) caused by tumor emboli, spinal cord complications, and intracranial second malignancies in PPB. Procedure. International PPB Registry and literature cases were evaluated for CNS events. Cerebral metastasis patients were evaluated for gender, side of origin of PPB, PPB Type, interval from diagnosis to metastasis, status of chest disease, treatment, and outcome. Standard statistical methods were used to calculate the cumulative probability of cerebral metastasis and survival following metastasis. Results. Thirty-nine cases of cerebral metastasis were identified in 5/53 Registry Type II cases, 15/44 Registry Type III cases, and 19/143 literature Type II/III cases. Metastases occurred 1-60, median 11.5 months after diagnosis. Chest disease was controlled in 50% of children at time of metastasis. The cumulative probability of cerebral metastasis by 5 years from diagnosis was 11 % for Type II patients (95%CI (confidence interval): 2-20%) and 54% for Type III patients (95%CI: 31-76%). Seven children survive cerebral metastasis. Other CNS complications were post-operative CVA (five cases), spinal cord invasion or compression (six), leptomeningeal disease (three), and second intracranial malignancies (two). Conclusions. Cerebral metastasis is more frequent in PPB than in other childhood sarcomas. Clinicians should screen for this complication. Diverse other CNS complications are less common and require careful diagnosis.

KW - Cerebral metastasis

KW - Cerebrovascular accidents

KW - Pleuropulmonary blastoma

KW - Second intracranial neoplasms

KW - Tumor embolism

UR - http://www.scopus.com/inward/record.url?scp=34547802219&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=34547802219&partnerID=8YFLogxK

U2 - 10.1002/pbc.20937

DO - 10.1002/pbc.20937

M3 - Article

C2 - 16807914

AN - SCOPUS:34547802219

VL - 49

SP - 266

EP - 273

JO - Pediatric Blood and Cancer

JF - Pediatric Blood and Cancer

SN - 1545-5009

IS - 3

ER -