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INFORM2 Exploratory Multinational Phase I/II Combination Study of Nivolumab and Entinostat in Children and Adolescents With Refractory High-risk Malignancies

ACTRN 12619001768189

Brief Summary

The aim of this clinical trial is to find out whether combination treatment with nivolumab and entinostat is safe and effective for children and adolescents with high-risk cancer.

Intervention/Treatment

  • Drug: Nivolumab
  • Drug: Entinostat

Inclusion Criteria

  • Children and adolescents with refractory/relapsed/progressive high-risk:
    • CNS tumours: medulloblastoma, ependymoma, ATRT, ETMR, paediatric high grade glioma (including DIPG) or other paediatric embryonal CNS tumours OR
    • solid tumours: neuroblastoma, nephroblastoma, rhabdoid tumour, embryonal or alveolar rhabdomyosarcoma or other embryonal small round blue cell tumours including paediatric type (bone) sarcoma OR
    • Children and adolescents with newly diagnosed high grade glioma (HGG) in the context of a constitutional mismatch repair deficiency syndrome after maximum safe surgical resection with no established standard of care treatment option with curative intention available.
  • No standard of care treatment available.
  • Age at registration greater than or equal to 2 years to less than or equal to 21 years.
  • Molecular analysis for biomarker identification (SNV load, high TILs or TLS positive, MYC/N amplification) in laboratories complying with DIN EN ISO/IEC 17025 or similar via INFORM molecular diagnostic platform or equivalently valid molecular pipeline.
  • Biomarker determined using whole exome sequencing (SNV load), IHC (high TILs or TLS positive) and whole genome sequencing (MYC/N amplification).
  • In case molecular analysis was not performed via INFORM molecular pipeline: transfer of molecular data (whole genome sequencing).
  • Time between biopsy/puncture/resection of the current refractory/relapsed/progressive tumour and registration less than or equal to 24 weeks.
  • Disease that is measurable as defined by RANO criteria or RECIST v1.1.
  • Life expectancy > 3 months, sufficient general condition score (Lansky greater than or equal to 70 or Karnofsky greater than or equal to 70). Transient states like infections requiring antibiotic treatments can be accepted, and also stable disabilities resulting from disease/surgery (hemiparesis, amputations etc.) can be accepted and will not be considered for Lansky/Karnofsky assessments.
  • Laboratory requirements:
    • Hematology: absolute granulocytes greater than or equal to 1.0 × 10^9/L (unsupported), platelets greater than or equal to 100 × 10^9/L, hemoglobin greater than or equal to 8 g/dL or greater than or equal to 4.96 nmol/L.
    • Biochemistry: Total bilirubin less than or equal to 1.5 x upper limit of normal (ULN) AST(SGOT) less than or equal to 3.0 x ULN, ALT(SGPT) less than or equal to 3.0 x ULN, serum creatinine less than or equal to 1.5 x ULN for age.
  • ECG: normal QTc interval according to Bazett formula < 440ms.
  • Patient is able to swallow oral study medication.
  • Ability of patient and/or legal representative(s) to understand the character and individual consequences of clinical trial.
  • Females of childbearing potential must have a negative serum or urine pregnancy test within 7 days prior to initiation of treatment. Sexually active women of childbearing potential must agree to use acceptable and appropriate contraception during the study and for at least 6 months after the last study treatment administration. Sexually active male patients must agree to use a condom during the study and for at least 7 months after the last study treatment administration.
  • Absence of any psychological, familial, sociological or geographical condition potentially hampering compliance with the study protocol and follow-up schedule; those conditions should be discussed with the patient before registration in the trial.
  • Before patient screening and registration, written informed consent, also concerning data and blood transfer, must be given according to ICH/GCP, and national/local regulations.
  • No prior therapy with the combination of immune checkpoint inhibitors and HDACi.
  • Phase I: molecular analysis performed and biomarker status known (mutational load, high TILs or TLS positive and MYC(N) amplification status).
  • Phase II: molecular analysis performed, biomarker status known (mutational load, high TILs or TLS positive and MYC(N) amplification status) and stratification according to the following criteria:
    • Group A: High mutational load (defined as > 100 somatic SNVs/exome) based on whole exome sequencing or
    • Group C: Focal MYC(N) amplification based on whole genome sequencing or ATRT-MYC subgroup or
    • Group E: High TILs or TLS positive (defined as cells per mm^2 > 600 or presence of tertiary lymphoid structure) based on IHC analysis

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