Epithelioid hemangioendothelioma in children: The European Pediatric Soft Tissue Sarcoma Study Group experience

Daniel Orbach; Max M Van Noesel; Bernadette Brennan; Nadège Corradini; Rita Alaggio; Myriam Ben Arush; Reineke A Schoot; Pablo Berlanga; Ilaria Zanetti; Lisa Lyngsie Hjalgrim; Federica Di Corti; Gema Ramirez; Michela Casanova; Andrea Ferrari

doi:10.1002/pbc.29882

Epithelioid hemangioendothelioma in children: The European Pediatric Soft Tissue Sarcoma Study Group experience

Daniel Orbach
, Max M Van Noesel
, Bernadette Brennan
, Nadège Corradini
, Rita Alaggio
, Myriam Ben Arush
, Reineke A Schoot
, Pablo Berlanga
, Ilaria Zanetti
, Lisa Lyngsie Hjalgrim
, Federica Di Corti
, Gema Ramirez
, Michela Casanova
, Andrea Ferrari

Research output: Contribution to journal › Article › Academic › peer-review

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Abstract

The European pediatric Soft tissue sarcoma Study Group analyzed all children with epithelioid hemangioendothelioma prospectively registered in the NRSTS-05 (EUDRACT 2005-001139-31) and in MTS-2008 (NCT00379457) studies: 10 patients with localized and one with metastatic disease. Median age was 14.3 years (range, 9.0-18.8). Local therapy was initial primary surgery in seven cases, and five patients received systemic therapy. No patients received radiotherapy. After a median follow-up of 50 months (range, 6-176) for living patients, nine patients remain alive off therapy and two died. Five-year progression free and overall survivals are, respectively, 77.1% (95% confidence interval [CI]: 34.5-93.9) and 74.1% (95% CI: 28.1-93.0).

Original language	English
Article number	e29882
Journal	Pediatric Blood & Cancer
Volume	69
Issue number	10
Early online date	16 Jul 2022
DOIs	https://doi.org/10.1002/pbc.29882
Publication status	Published - Oct 2022

Keywords

children
hemangioendothelioma
soft tissue sarcoma
surgery

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10.1002/pbc.29882Licence: CC BY-NC-ND

Pediatric Blood Cancer - 2022 - Orbach - Epithelioid hemangioendothelioma in children The European Pediatric Soft TissueFinal published version, 269 KBLicence: CC BY-NC-ND

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Orbach, D., Van Noesel, M. M., Brennan, B., Corradini, N., Alaggio, R., Ben Arush, M., Schoot, R. A., Berlanga, P., Zanetti, I., Hjalgrim, L. L., Di Corti, F., Ramirez, G., Casanova, M., & Ferrari, A. (2022). Epithelioid hemangioendothelioma in children: The European Pediatric Soft Tissue Sarcoma Study Group experience. Pediatric Blood & Cancer, 69(10), Article e29882. https://doi.org/10.1002/pbc.29882

@article{f973e149cdb74e45b9393adb5e7973ed,

title = "Epithelioid hemangioendothelioma in children: The European Pediatric Soft Tissue Sarcoma Study Group experience",

abstract = "The European pediatric Soft tissue sarcoma Study Group analyzed all children with epithelioid hemangioendothelioma prospectively registered in the NRSTS-05 (EUDRACT 2005-001139-31) and in MTS-2008 (NCT00379457) studies: 10 patients with localized and one with metastatic disease. Median age was 14.3 years (range, 9.0-18.8). Local therapy was initial primary surgery in seven cases, and five patients received systemic therapy. No patients received radiotherapy. After a median follow-up of 50 months (range, 6-176) for living patients, nine patients remain alive off therapy and two died. Five-year progression free and overall survivals are, respectively, 77.1\% (95\% confidence interval [CI]: 34.5-93.9) and 74.1\% (95\% CI: 28.1-93.0).",

keywords = "children, hemangioendothelioma, soft tissue sarcoma, surgery",

author = "Daniel Orbach and \{Van Noesel\}, \{Max M\} and Bernadette Brennan and Nad{\`e}ge Corradini and Rita Alaggio and \{Ben Arush\}, Myriam and Schoot, \{Reineke A\} and Pablo Berlanga and Ilaria Zanetti and Hjalgrim, \{Lisa Lyngsie\} and \{Di Corti\}, Federica and Gema Ramirez and Michela Casanova and Andrea Ferrari",

note = "Funding Information: This study is in memory of Dr. S. Schiffler, member of the EpSSG NRSTS committee. The authors want to thank Dr. B. Coppadoro for her help in this project. Publisher Copyright: {\textcopyright} 2022 The Authors. Pediatric Blood \& Cancer published by Wiley Periodicals, Inc.",

year = "2022",

month = oct,

doi = "10.1002/pbc.29882",

language = "English",

volume = "69",

journal = "Pediatric Blood \& Cancer",

issn = "1545-5009",

publisher = "Wiley-Liss Inc.",

number = "10",

}

Orbach, D, Van Noesel, MM, Brennan, B, Corradini, N, Alaggio, R, Ben Arush, M, Schoot, RA, Berlanga, P, Zanetti, I, Hjalgrim, LL, Di Corti, F, Ramirez, G, Casanova, M & Ferrari, A 2022, 'Epithelioid hemangioendothelioma in children: The European Pediatric Soft Tissue Sarcoma Study Group experience', Pediatric Blood & Cancer, vol. 69, no. 10, e29882. https://doi.org/10.1002/pbc.29882

TY - JOUR

T1 - Epithelioid hemangioendothelioma in children

T2 - The European Pediatric Soft Tissue Sarcoma Study Group experience

AU - Orbach, Daniel

AU - Van Noesel, Max M

AU - Brennan, Bernadette

AU - Corradini, Nadège

AU - Alaggio, Rita

AU - Ben Arush, Myriam

AU - Schoot, Reineke A

AU - Berlanga, Pablo

AU - Zanetti, Ilaria

AU - Hjalgrim, Lisa Lyngsie

AU - Di Corti, Federica

AU - Ramirez, Gema

AU - Casanova, Michela

AU - Ferrari, Andrea

N1 - Funding Information: This study is in memory of Dr. S. Schiffler, member of the EpSSG NRSTS committee. The authors want to thank Dr. B. Coppadoro for her help in this project. Publisher Copyright: © 2022 The Authors. Pediatric Blood & Cancer published by Wiley Periodicals, Inc.

PY - 2022/10

Y1 - 2022/10

N2 - The European pediatric Soft tissue sarcoma Study Group analyzed all children with epithelioid hemangioendothelioma prospectively registered in the NRSTS-05 (EUDRACT 2005-001139-31) and in MTS-2008 (NCT00379457) studies: 10 patients with localized and one with metastatic disease. Median age was 14.3 years (range, 9.0-18.8). Local therapy was initial primary surgery in seven cases, and five patients received systemic therapy. No patients received radiotherapy. After a median follow-up of 50 months (range, 6-176) for living patients, nine patients remain alive off therapy and two died. Five-year progression free and overall survivals are, respectively, 77.1% (95% confidence interval [CI]: 34.5-93.9) and 74.1% (95% CI: 28.1-93.0).

AB - The European pediatric Soft tissue sarcoma Study Group analyzed all children with epithelioid hemangioendothelioma prospectively registered in the NRSTS-05 (EUDRACT 2005-001139-31) and in MTS-2008 (NCT00379457) studies: 10 patients with localized and one with metastatic disease. Median age was 14.3 years (range, 9.0-18.8). Local therapy was initial primary surgery in seven cases, and five patients received systemic therapy. No patients received radiotherapy. After a median follow-up of 50 months (range, 6-176) for living patients, nine patients remain alive off therapy and two died. Five-year progression free and overall survivals are, respectively, 77.1% (95% confidence interval [CI]: 34.5-93.9) and 74.1% (95% CI: 28.1-93.0).

KW - children

KW - hemangioendothelioma

KW - soft tissue sarcoma

KW - surgery

UR - https://www.scopus.com/pages/publications/85134177135

U2 - 10.1002/pbc.29882

DO - 10.1002/pbc.29882

M3 - Article

C2 - 35841307

SN - 1545-5009

VL - 69

JO - Pediatric Blood & Cancer

JF - Pediatric Blood & Cancer

IS - 10

M1 - e29882

ER -

Epithelioid hemangioendothelioma in children: The European Pediatric Soft Tissue Sarcoma Study Group experience

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