Results of a phase II pilot study of moderate dose radiotherapy for inoperable desmoid-type fibromatosis-an EORTC STBSG and ROG study (EORTC 62991-22998)

Keus, R.B.; Nout, R.A.; Blay, J-Y.; de Jong, J.M.; Hennig, I.; Saran, F.; Hartmann, J.T.; Sunyach, M.P.; Gwyther, S.J.; Ouali, M.; Kirkpatrick, A.; Poortmans, P.M.; Hogendoorn, P.C.W.; van der Graaf, W.T.A.

doi:10.1093/ANNONC/MDT254

Title

Results of a phase II pilot study of moderate dose radiotherapy for inoperable desmoid-type fibromatosis-an EORTC STBSG and ROG study (EORTC 62991-22998)

Author

Keus, R.B.

Nout, R.A.

Blay, J-Y.

de Jong, J.M.

Hennig, I.

Saran, F.

Hartmann, J.T.

Sunyach, M.P.

Gwyther, S.J.

Ouali, M.

Kirkpatrick, A.

Poortmans, P.M.

Hogendoorn, P.C.W.

van der Graaf, W.T.A.

Abstract

To determine the activity of radiotherapy in patients with inoperable desmoid-type fibromatosis (DF) a multicenter prospective phase II trial was carried out. Patients with inoperable progressive disease of primary, recurrent or incompletely resected lesions received a dose of 56 Gy in 28 fractions. Follow-up MRI studies were carried out every 3 months for 2 years and thereafter every 6 months. The primary end point was local control rate at 3 years, estimated by a nonparametric method for interval-censored survival data. Secondary end points were objective tumor response, acute and late toxic effect. Forty-four patients (27 F/17 M) were enrolled from 2001 to 2008. Median age was 39.5 years. Main tumor sites included trunk 15 (34.1%) and extremities 27 (61.3%). Median follow-up was 4.8 years. The 3-year local control rate was 81.5% (90% one-sided confidence interval 74% to 100%). Best overall response during the first 3 years was complete response (CR) 6 (13.6%), partial response (PR) 16 (36.4%), stable disease 18 (40.9%), progressive disease 3 (6.8%) and nonassessable 1 (2.3%). Five patients developed new lesions. After 3 years, the response further improved in three patients: (CR 2, PR 1). Acute grade 3 side-effects were limited to skin, mucosal membranes and pain. Late toxic effect consisted of mild edema in 10 patients. Moderate dose radiotherapy is an effective treatment of patients with DF. Response after radiation therapy is slow with continuing regression seen even after 3 years.

Language

English

Source (journal)

Annals of oncology / European Society for Medical Oncology. - Amsterdam

Publication

Amsterdam : 2013

ISSN

0923-7534

DOI

10.1093/ANNONC/MDT254

Volume/pages

24 :10 (2013) , p. 2672-2676