Sunitinib treatment in pediatric patients with advanced GIST following failure of imatinib

Katherine A. Janeway, Karen H. Albritton, Annick D. Van Den Abbeele, Gina Z. D'Amato, Paolo Pedrazzoli, Salvatore Siena, Joel Picus, James E. Butrynski, Marcus Schlemmer, Michael C. Heinrich, George D. Demetri

Research output: Contribution to journalArticle

99 Citations (Scopus)

Abstract

Background. Sunitinib inhibits KIT and other members of the split-kinase-domain family of receptor tyrosine kinases. Sunitinib prolongs survival in adult patients with imatinib-resistant gastrointestinal stromal tumor (GIST). We report the experience with sunitinib in pediatric patients with advanced GIST following failure of imatinib. Procedure. Sunitinib therapy was provided through a treatment-use protocol. Patients were 10-17 years old at enrollment. All patients had GIST resistant to imatinib therapy. Sunitinib was administered daily for 4 weeks in 6-week treatment cycles. KIT and platelet-derived growth factor receptor alpha (PDGFRA) genotyping of tumor tissue were performed. Results. One patient achieved a partial response, five patients had stable disease and one patient had progressive disease on sunitinib. The duration of disease stabilization was between 7 and 21+ months, with a mean of 15 months. Time to tumor progression was longer on sunitinib than on prior imatinib treatment for five of six patients. Two patients experienced grade 3 adverse events. All other adverse events were grade 1-2. None of the five patients tested had mutations in KIT or PDGFRA. Conclusion. Sunitinib treatment was associated with substantial initial antitumor activity and acceptable tolerability in this group of pediatric patients with imatinib-resistant GIST.

Original languageEnglish
Pages (from-to)767-771
Number of pages5
JournalPediatric Blood and Cancer
Volume52
Issue number7
DOIs
Publication statusPublished - Jul 1 2009

Fingerprint

Gastrointestinal Stromal Tumors
Pediatrics
Therapeutics
Platelet-Derived Growth Factor alpha Receptor
Imatinib Mesylate
sunitinib
Receptor Protein-Tyrosine Kinases
Clinical Protocols
Neoplasms
Phosphotransferases

Keywords

  • Clinical trials
  • Drug resistance
  • New agents
  • Pediatric oncology
  • Soft tissue sarcoma

ASJC Scopus subject areas

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

Cite this

Janeway, K. A., Albritton, K. H., Van Den Abbeele, A. D., D'Amato, G. Z., Pedrazzoli, P., Siena, S., ... Demetri, G. D. (2009). Sunitinib treatment in pediatric patients with advanced GIST following failure of imatinib. Pediatric Blood and Cancer, 52(7), 767-771. https://doi.org/10.1002/pbc.21909

Sunitinib treatment in pediatric patients with advanced GIST following failure of imatinib. / Janeway, Katherine A.; Albritton, Karen H.; Van Den Abbeele, Annick D.; D'Amato, Gina Z.; Pedrazzoli, Paolo; Siena, Salvatore; Picus, Joel; Butrynski, James E.; Schlemmer, Marcus; Heinrich, Michael C.; Demetri, George D.

In: Pediatric Blood and Cancer, Vol. 52, No. 7, 01.07.2009, p. 767-771.

Research output: Contribution to journalArticle

Janeway, KA, Albritton, KH, Van Den Abbeele, AD, D'Amato, GZ, Pedrazzoli, P, Siena, S, Picus, J, Butrynski, JE, Schlemmer, M, Heinrich, MC & Demetri, GD 2009, 'Sunitinib treatment in pediatric patients with advanced GIST following failure of imatinib', Pediatric Blood and Cancer, vol. 52, no. 7, pp. 767-771. https://doi.org/10.1002/pbc.21909
Janeway, Katherine A. ; Albritton, Karen H. ; Van Den Abbeele, Annick D. ; D'Amato, Gina Z. ; Pedrazzoli, Paolo ; Siena, Salvatore ; Picus, Joel ; Butrynski, James E. ; Schlemmer, Marcus ; Heinrich, Michael C. ; Demetri, George D. / Sunitinib treatment in pediatric patients with advanced GIST following failure of imatinib. In: Pediatric Blood and Cancer. 2009 ; Vol. 52, No. 7. pp. 767-771.
@article{ed8fad8ea72546a58c80e0f0f0fb27e2,
title = "Sunitinib treatment in pediatric patients with advanced GIST following failure of imatinib",
abstract = "Background. Sunitinib inhibits KIT and other members of the split-kinase-domain family of receptor tyrosine kinases. Sunitinib prolongs survival in adult patients with imatinib-resistant gastrointestinal stromal tumor (GIST). We report the experience with sunitinib in pediatric patients with advanced GIST following failure of imatinib. Procedure. Sunitinib therapy was provided through a treatment-use protocol. Patients were 10-17 years old at enrollment. All patients had GIST resistant to imatinib therapy. Sunitinib was administered daily for 4 weeks in 6-week treatment cycles. KIT and platelet-derived growth factor receptor alpha (PDGFRA) genotyping of tumor tissue were performed. Results. One patient achieved a partial response, five patients had stable disease and one patient had progressive disease on sunitinib. The duration of disease stabilization was between 7 and 21+ months, with a mean of 15 months. Time to tumor progression was longer on sunitinib than on prior imatinib treatment for five of six patients. Two patients experienced grade 3 adverse events. All other adverse events were grade 1-2. None of the five patients tested had mutations in KIT or PDGFRA. Conclusion. Sunitinib treatment was associated with substantial initial antitumor activity and acceptable tolerability in this group of pediatric patients with imatinib-resistant GIST.",
keywords = "Clinical trials, Drug resistance, New agents, Pediatric oncology, Soft tissue sarcoma",
author = "Janeway, {Katherine A.} and Albritton, {Karen H.} and {Van Den Abbeele}, {Annick D.} and D'Amato, {Gina Z.} and Paolo Pedrazzoli and Salvatore Siena and Joel Picus and Butrynski, {James E.} and Marcus Schlemmer and Heinrich, {Michael C.} and Demetri, {George D.}",
year = "2009",
month = "7",
day = "1",
doi = "10.1002/pbc.21909",
language = "English",
volume = "52",
pages = "767--771",
journal = "Pediatric Blood and Cancer",
issn = "1545-5009",
publisher = "John Wiley and Sons Inc.",
number = "7",

}

TY - JOUR

T1 - Sunitinib treatment in pediatric patients with advanced GIST following failure of imatinib

AU - Janeway, Katherine A.

AU - Albritton, Karen H.

AU - Van Den Abbeele, Annick D.

AU - D'Amato, Gina Z.

AU - Pedrazzoli, Paolo

AU - Siena, Salvatore

AU - Picus, Joel

AU - Butrynski, James E.

AU - Schlemmer, Marcus

AU - Heinrich, Michael C.

AU - Demetri, George D.

PY - 2009/7/1

Y1 - 2009/7/1

N2 - Background. Sunitinib inhibits KIT and other members of the split-kinase-domain family of receptor tyrosine kinases. Sunitinib prolongs survival in adult patients with imatinib-resistant gastrointestinal stromal tumor (GIST). We report the experience with sunitinib in pediatric patients with advanced GIST following failure of imatinib. Procedure. Sunitinib therapy was provided through a treatment-use protocol. Patients were 10-17 years old at enrollment. All patients had GIST resistant to imatinib therapy. Sunitinib was administered daily for 4 weeks in 6-week treatment cycles. KIT and platelet-derived growth factor receptor alpha (PDGFRA) genotyping of tumor tissue were performed. Results. One patient achieved a partial response, five patients had stable disease and one patient had progressive disease on sunitinib. The duration of disease stabilization was between 7 and 21+ months, with a mean of 15 months. Time to tumor progression was longer on sunitinib than on prior imatinib treatment for five of six patients. Two patients experienced grade 3 adverse events. All other adverse events were grade 1-2. None of the five patients tested had mutations in KIT or PDGFRA. Conclusion. Sunitinib treatment was associated with substantial initial antitumor activity and acceptable tolerability in this group of pediatric patients with imatinib-resistant GIST.

AB - Background. Sunitinib inhibits KIT and other members of the split-kinase-domain family of receptor tyrosine kinases. Sunitinib prolongs survival in adult patients with imatinib-resistant gastrointestinal stromal tumor (GIST). We report the experience with sunitinib in pediatric patients with advanced GIST following failure of imatinib. Procedure. Sunitinib therapy was provided through a treatment-use protocol. Patients were 10-17 years old at enrollment. All patients had GIST resistant to imatinib therapy. Sunitinib was administered daily for 4 weeks in 6-week treatment cycles. KIT and platelet-derived growth factor receptor alpha (PDGFRA) genotyping of tumor tissue were performed. Results. One patient achieved a partial response, five patients had stable disease and one patient had progressive disease on sunitinib. The duration of disease stabilization was between 7 and 21+ months, with a mean of 15 months. Time to tumor progression was longer on sunitinib than on prior imatinib treatment for five of six patients. Two patients experienced grade 3 adverse events. All other adverse events were grade 1-2. None of the five patients tested had mutations in KIT or PDGFRA. Conclusion. Sunitinib treatment was associated with substantial initial antitumor activity and acceptable tolerability in this group of pediatric patients with imatinib-resistant GIST.

KW - Clinical trials

KW - Drug resistance

KW - New agents

KW - Pediatric oncology

KW - Soft tissue sarcoma

UR - http://www.scopus.com/inward/record.url?scp=67650472798&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=67650472798&partnerID=8YFLogxK

U2 - 10.1002/pbc.21909

DO - 10.1002/pbc.21909

M3 - Article

C2 - 19326424

AN - SCOPUS:67650472798

VL - 52

SP - 767

EP - 771

JO - Pediatric Blood and Cancer

JF - Pediatric Blood and Cancer

SN - 1545-5009

IS - 7

ER -