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Evaluation of Clinical Impact of Interruption VS Maintenance of AI in Patients with Locally Advanced/ Metastatic Low Grade Endometrial Stromal Sarcoma (LGESS)

Phase 2
Recruiting
Conditions
Low Grade Endometrial Stromal Sarcoma
Interventions
Drug: Aromatase Inhibitors
Registration Number
NCT03624244
Lead Sponsor
Centre Leon Berard
Brief Summary

The primary objective is to compare the progression-free survival (PFS) between aromatase inhibitors interruption and aromatase inhibitors maintenance strategies in patients with a locally advanced or metastatic Low Grade Endometrial Stromal Sarcoma (LGESS).

Detailed Description

Uterine sarcomas are rare tumors with an incidence of 1.7/100 000 women per year, including 20% of endometrial stromal sarcomas (ESS). Patients with low grade ESS (LGESS) have a good prognosis with a 5-year overall survival rates ranging from 66 to 98%, depending on the stage of the disease.

Majority of LGESS report estrogen receptor (ER) and/or progesterone receptor positive and a chromosomal translocation with JAZF1-SUZ12.

Based on the current European Society of Medical Oncology (ESMO)guidelines, the standard treatment for patients with early/non metastatic ESS is total hysterectomy plus or less bilateral salpingo-oophorectomy. The use of hormonal therapy (HT) for advanced or metastatic disease is recommended based on retrospective data from small series providing evidence that HT have an anti-tumor activity on LGESS. HT includes aromatase inhibitors (AI), progestins and gonadotrophin-releasing hormone.

Very few data are available in this rare disease, but retrospective analyses show that AI may provide response rates of 46 to 67% in metastatic LGESS patients (7% complete response, 60% partial response), with a mean duration of response of 24 months.

Even if AI are effective and well tolerated, chronical mild to moderate (grade 1-2) side-effects (arthritis, hot-flashes, osteoporosis, hypercholesterolemia, cardiac events) have a negative impact on patient's well-being because of the treatment long term duration and need to be balanced in such long term survival.

To date, the question of the optimal duration of HT in LGESS is still pending. The investigator propose an open-label, randomized, multicenter phase II study aiming at determining the feasibility of interruption of AI in patients with locally advanced or metastatic LGESS after long term stabilization or response to AI. The study will use a sequential bayesian design allowing for continuous monitoring of the main efficacy outcome, thus leading to a smaller more informative trial, and specifically tied to decision making. This design is particularly suited to characterize efficacy signals in the context of a very rare pathology. Moreover JAZF1-JJAZ1 fusion gene is not identified in all LGESS.

Ancillary studies will provide precious data aiming at:

* Identifying predictive factors of prolonged response to HT or late resistance (Next Generation Sequencing and Comparative Genome Hybridization).

* Evaluating sociobehavioral (only for French sites) of patients by following questionnaire: Zimbardo Time Perspective Inventory (ZTPI) , Functional, Communicative and Critical Health Literacy/ 14-item Health Literacy Scale (FCCHL/HLS14), VICAN, Fear of Cancer Recurrence (FCR) and Patient-Generated Index (PGI).

Recruitment & Eligibility

Status
RECRUITING
Sex
Female
Target Recruitment
40
Inclusion Criteria
  • Age≥18 years;
  • Histological confirmation of low grade ESS;
  • Locally advanced or metastatic disease at diagnosis or patient experiencing a tumor effraction during hysterectomy;
  • Treatment with aromatase inhibitors (Anastrozole or Exemestane or Letrozole ) initiated either: for at least 24 months (in patients with no residual disease or non-measurable disease at the last AI initiation) OR for at least 36 months (in patients with measurable disease at the last AI initiation);
  • Disease must be controlled at the time of the randomisation (objective response or stable disease) by the aromatase inhibitor initiated either for at least 24 or 36 months
  • Eastern Cooperative Oncology Group (ECOG) Performance Status (PS) ≤ 2
  • Covered by a medical insurance;
  • Signed informed consent prior to any study-specific procedure.
Exclusion Criteria
  • Pregnant or breastfeeding woman;
  • Patient concurrently using other approved or investigational antineoplastic agents;
  • Major concurrent disease affecting cardiovascular system, liver, kidneys, hematopoietic system or else considered as clinically important by the investigator and that could be incompatible with patient's participation in this trial or would likely interfere with study procedures or results;
  • Prior history of malignancies other than study disease (except for basal cell or squamous cell carcinoma of the skin or carcinoma in situ of the cervix) unless the patient has been free of the disease for at least 3 years;
  • Patients using prohibited concomitant and/or concurrent medications
  • Contra-indication according to SmPCs.
  • Patient requiring tutorship or curatorship.

Study & Design

Study Type
INTERVENTIONAL
Study Design
PARALLEL
Arm && Interventions
GroupInterventionDescription
Maintenance of aromatase inhibitorsAromatase InhibitorsMaintenance of aromatase inhibitors
Interruption of aromatase inhibitorsAromatase InhibitorsInterruption of aromatase inhibitors until progression disease. At disease progression, AI can be reintroduced.
Primary Outcome Measures
NameTimeMethod
Progression free survivalFrom date of randomization until the date of first documented progression or date of death from any cause, whichever came first, assessed up to 60 months

Progression free survival

Secondary Outcome Measures
NameTimeMethod
Objective response rate after reintroduction of AI in the experimental armFrom the date of AI reintroduction in the experimental arm to the date of subsequent progression or date of death due to any cause, whichever came first, assessed up to 60 months

Proportion of patients with a best overall response of Partial Response (PR) or Complete Response (CR) after AI reintroduction in the experimental arm

Progression free survival after reintroduction of AI in the experimental armFrom the date of AI reintroduction in the experimental arm to the date of subsequent progression or date of death due to any cause, whichever came first, assessed up to 60 months

Progression free survival after reintroduction of AI in the experimental arm

Quality of Life using European Organization for Research and Treatment of Cancer (EORTC) Quality of Life Questionnaire Core 30 (QLQ-C30)Every 6 months until the 36th month for each patient

Quality of Life using EORTC QLQ-C30 questionnaire. 64 questions related to cancer impact on health and daily activities composed this questionnaire. Each item has to be graded from 1 to 4 ( 1 = not at all; 4 = very much). More the score is high, worst the quality of life is.

Overall survivalFrom date of randomization to death due to any cause, assessed up to 60 months

Overall survival

Duration of response to AI after reintroductionFrom the date of first objective response following the reintroduction of AI to the date of the first subsequent documented radiological progression or death due to any cause, whichever came first, assessed up to 60 months

Duration of response to AI after reintroduction

Time to first subsequent chemotherapy/treatment or deathFrom date of randomization to the earliest date of chemotherapy/treatment start date following study treatment discontinuation, or death due to any cause, whichever came first, assessed up to 60 months

Time to first subsequent chemotherapy/treatment or death

Incidence of Treatment-Emergent Adverse EventsFrom date of randomization to follow-up visit Month 36 or death due to any cause, whichever came first, assessed up to 60 months

Safety and Tolerability assessed according to the NCI-CTC AE version 5

Trial Locations

Locations (21)

CHU Tours

🇫🇷

Tours, France

CHU Besançon

🇫🇷

Besançon, France

Insitut Bergonié

🇫🇷

Bordeaux, France

Centre Jean Perrin

🇫🇷

Clermont-Ferrand, France

Centre François Baclesse

🇫🇷

Caen, France

Centre Oscar Lambret

🇫🇷

Lille, France

CHU Dupuytren

🇫🇷

Limoges, France

Centre Léon Bérard

🇫🇷

Lyon, France

Hopital La Timone

🇫🇷

Marseille, France

Institut de Cancérologie de Montpellier

🇫🇷

Montpellier, France

Institut Paoli Calmette

🇫🇷

Marseille, France

Centre Antoine Lacassagne

🇫🇷

Nice, France

Hopital Pitié Salpétrière

🇫🇷

Paris, France

Centre Henri Becquerel

🇫🇷

Rouen, France

Insitut Curie

🇫🇷

Paris, France

Institut Godinot

🇫🇷

Reims, France

AP-HP Hopîtal Cochin

🇫🇷

Paris, France

CHUSE

🇫🇷

Saint-Priest-en-Jarez, France

Hopital Privé de la Loire

🇫🇷

Saint-Étienne, France

ICO Centre René Gauducheau

🇫🇷

Saint-Herblain, France

Institut Gustave Roussy

🇫🇷

Villejuif, France

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