Can we cure patients with abdominal Desmoplastic Small Round Cell Tumor? Results of a retrospective multicentric study on 100 patients.


Journal

Surgical oncology
ISSN: 1879-3320
Titre abrégé: Surg Oncol
Pays: Netherlands
ID NLM: 9208188

Informations de publication

Date de publication:
Jun 2019
Historique:
received: 07 08 2018
revised: 18 12 2018
accepted: 08 04 2019
entrez: 15 6 2019
pubmed: 15 6 2019
medline: 19 12 2019
Statut: ppublish

Résumé

Despite being associated with a very poor prognosis, long-term survivors across all series of Desmoplastic Small Round Cell Tumor (DSRCT) have been reported. To analyze patients 'characteristics associated with a prolonged survival after DSRCT diagnosis. All consecutive patients treated for DSRCT in nine French expert centers between 1991 and 2018 were retrospectively analyzed. Patients with a follow-up of less than 2 years were excluded and cure defined as being disease-free at least 5 years. 100 pts were identified (median age 25 years, 89% male). 27 had distant metastases at diagnosis and 80 pts underwent upfront chemotherapy (CT). 71 pts were operated, 20 pts without prior CT). Surgery was macroscopically complete (CC0/1) in 50 pts. Hyperthermic intraperitoneal Chemotherapy (HIPEC) was administered during surgery in 15 pts 54 pts had postoperative CT and 26 pts had postoperative whole abdomino-pelvic RT (WAP-RT). After a median follow-up of 103 months (range 23-311), the median overall survival (OS) was 25 months. The 1- year, 3-year and 5-year OS rates were 90%, 35% and 4% respectively. 5 patients were considered cured after a median disease-free interval of 100 months (range 22-139). Predictive factors of cure were female sex (HR = 0.49, p = 0.014), median PCI<12 (HR = 0.32, p = 0.0004), MD Anderson stage I (HR = 0.25, p < 0.0001), CC0/1 (HR = 0.34, p < 0.0001), and WAP-RT (HR = 0.36, p = 0.00013). HIPEC did not statistically improve survival. Cure in DSRCT is possible in 5% of patients and is best achieved combining systemic chemotherapy, complete cytoreductive surgery and WAP-RT. Despite aggressive treatment, recurrence is common and targeted therapies are urgently needed.

Sections du résumé

BACKGROUND BACKGROUND
Despite being associated with a very poor prognosis, long-term survivors across all series of Desmoplastic Small Round Cell Tumor (DSRCT) have been reported.
AIM OF THE STUDY OBJECTIVE
To analyze patients 'characteristics associated with a prolonged survival after DSRCT diagnosis.
METHODS METHODS
All consecutive patients treated for DSRCT in nine French expert centers between 1991 and 2018 were retrospectively analyzed. Patients with a follow-up of less than 2 years were excluded and cure defined as being disease-free at least 5 years.
RESULTS RESULTS
100 pts were identified (median age 25 years, 89% male). 27 had distant metastases at diagnosis and 80 pts underwent upfront chemotherapy (CT). 71 pts were operated, 20 pts without prior CT). Surgery was macroscopically complete (CC0/1) in 50 pts. Hyperthermic intraperitoneal Chemotherapy (HIPEC) was administered during surgery in 15 pts 54 pts had postoperative CT and 26 pts had postoperative whole abdomino-pelvic RT (WAP-RT). After a median follow-up of 103 months (range 23-311), the median overall survival (OS) was 25 months. The 1- year, 3-year and 5-year OS rates were 90%, 35% and 4% respectively. 5 patients were considered cured after a median disease-free interval of 100 months (range 22-139). Predictive factors of cure were female sex (HR = 0.49, p = 0.014), median PCI<12 (HR = 0.32, p = 0.0004), MD Anderson stage I (HR = 0.25, p < 0.0001), CC0/1 (HR = 0.34, p < 0.0001), and WAP-RT (HR = 0.36, p = 0.00013). HIPEC did not statistically improve survival.
CONCLUSION CONCLUSIONS
Cure in DSRCT is possible in 5% of patients and is best achieved combining systemic chemotherapy, complete cytoreductive surgery and WAP-RT. Despite aggressive treatment, recurrence is common and targeted therapies are urgently needed.

Identifiants

pubmed: 31196472
pii: S0960-7404(18)30308-6
doi: 10.1016/j.suronc.2019.04.002
pii:
doi:

Types de publication

Journal Article Multicenter Study

Langues

eng

Sous-ensembles de citation

IM

Pagination

107-112

Informations de copyright

Copyright © 2019. Published by Elsevier Ltd.

Auteurs

C Honoré (C)

Department of Surgical Oncology, Gustave Roussy Cancer Campus, Villejuif, France. Electronic address: Charles.HONORE@gustaveroussy.fr.

J B Delhorme (JB)

Department of General and Digestive Surgery, Hautepierre Hospital, Strasbourg University, Strasbourg, France.

E Nassif (E)

Department of Cancer Medicine, Gustave Roussy Cancer Campus, Villejuif, France.

M Faron (M)

Department of Surgical Oncology, Gustave Roussy Cancer Campus, Villejuif, France.

G Ferron (G)

Department of Surgery, Claudius Régaud Institute (IUCT), Toulouse, France.

E Bompas (E)

Department of Medical Oncology, West Cancer Institute (ICO), Nantes, France.

O Glehen (O)

Department of Surgical Oncology, Lyon Civil Hospices, South Lyon University Hospital Center, Lyon, France.

A Italiano (A)

Department of Medical Oncology, Bergonié Institute, Bordeaux, France.

F Bertucci (F)

Department of Medical Oncology, Paoli-Calmettes Institute, Aix-Marseille University, Marseille, France.

D Orbach (D)

SIREDO Oncology Center (Care, Innovation and Research for Children, Adolescents and Young Adults with Cancer), Institut Curie, PSL University, Paris, France.

M Pocard (M)

Department of Digestive and Oncological Surgery, Lariboisière University Hospital, Paris, France.

F Quenet (F)

Department of Surgical Oncology, Regional Cancer Institute of Montpellier (ICM), Montpellier, France.

J Y Blay (JY)

Department of Medical Oncology, Leon Bérard Center, Lyon, France.

S Carrere (S)

Department of Surgical Oncology, Regional Cancer Institute of Montpellier (ICM), Montpellier, France.

C Chevreau (C)

Department of Medical Oncology, Claudius Régaud Institute (IUCT), Toulouse, France.

O Mir (O)

Department of Ambulatory Care, Gustave Roussy Cancer Campus, Villejuif, France.

A Le Cesne (A)

Department of Cancer Medicine, Gustave Roussy Cancer Campus, Villejuif, France.

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