scholarly journals Ketogenic diet treatment in diffuse intrinsic pontine glioma in children: Retrospective analysis of feasibility, safety, and survival data

2021 ◽  
Author(s):  
Alexandre Perez ◽  
Elles Louw ◽  
Janak Nathan ◽  
Moatasem El‐Ayadi ◽  
Hadrien Golay ◽  
...  
Keyword(s):  
Retrospective Analysis ◽  
Ketogenic Diet ◽  
Survival Data ◽  
Diffuse Intrinsic Pontine Glioma ◽  
Pontine Glioma ◽  
Diet Treatment

scholarly journals Ketogenic diet treatment in recurrent diffuse intrinsic pontine glioma in children: A safety and feasibility study

2018 ◽  
Vol 66 (3) ◽  
pp. e27561 ◽  
Author(s):  
Elles J.T.M. van der Louw ◽  
Roel E. Reddingius ◽  
Joanne F. Olieman ◽  
Rinze F. Neuteboom ◽  
Coriene E. Catsman-Berrevoets
Keyword(s):  
Ketogenic Diet ◽  
Feasibility Study ◽  
Diffuse Intrinsic Pontine Glioma ◽  
Pontine Glioma ◽  
Diet Treatment

Comment on: Ketogenic diet treatment in recurrent diffuse intrinsic pontine glioma in children: A safety and feasibility study

2019 ◽  
Vol 66 (7) ◽  
pp. e27664 ◽  
Author(s):  
Alexandre Perez ◽  
Laura Merlini ◽  
Moatasem El‐Ayadi ◽  
Christian Korff ◽  
Marc Ansari ◽  
...  
Keyword(s):  
Ketogenic Diet ◽  
Feasibility Study ◽  
Diffuse Intrinsic Pontine Glioma ◽  
Pontine Glioma ◽  
Diet Treatment

scholarly journals DIPG-25. KETOGENIC DIET IN DIFFUSE INTRINSIC PONTINE GLIOMA IN CHILDREN: A RETROSPECTIVE STUDY INVESTIGATING THE FEASIBILITY

2020 ◽  
Vol 22 (Supplement_3) ◽  
pp. iii291-iii292
Author(s):  
Alexandre Perez ◽  
Janak Nathan ◽  
Moatasem El-Ayadi ◽  
Christian Korff ◽  
Marc Ansari ◽  
...  
Keyword(s):  
Ketogenic Diet ◽  
Controlled Trial ◽  
Diffuse Intrinsic Pontine Glioma ◽  
Inclusion Criteria ◽  
Pontine Glioma ◽  
Randomized Controlled ◽  
Small Patient Population ◽  
Small Patient ◽  
The Impact ◽  
Aggressive Clinical Behavior

Abstract PURPOSE Diffuse Intrinsic Pontine Glioma (DIPG) is one of the most devastating diseases amongst children with cancer, thus novel strategies are urgently needed. We aimed to retrospectively evaluate the feasibility of the carbohydrate restricted ketogenic diet (KD) in DIPG patients. METHODS Searches of MEDLINE and Embase identified four publications meeting the inclusion criteria (diagnosis of DIPG and exposition to a KD ≥ 3 months). One additional case was identified by contact with experts. The minimal feasibility criteria were defined as the ability to use the KD for ≥ 3 months. Individual patient data were extracted from the publications or obtained from investigators. RESULTS Five patients (males, n=3; median age 4.4 years; range, 2.5–17 years) met the inclusion criteria (one patient – identified and not included - was on KD < 3 months due to disease progression). Further feasibility analyses showed a duration of the KD of ≥ 3 months and less than 7 months (n=2), > 7 months and less than 1 year (n= 2), and two years (n=1), respectively. CONCLUSION These results – based on a small patient population – suggest that the KD appears to be a feasible treatment option for children with DIPG. The potential duration of the KD is limited by the aggressive clinical behavior of DIPG. The safety analysis is currently being retrospectively assessed. These data should encourage further studies on a larger scale; ideally assessing the impact of the KD in DIPG patients in a randomized controlled trial.

Retrospective analysis on the consistency of MRI features with histological and molecular markers in diffuse intrinsic pontine glioma (DIPG)

2019 ◽  
Vol 36 (4) ◽  
pp. 697-704
Author(s):  
Marzia Giagnacovo ◽  
Manila Antonelli ◽  
Veronica Biassoni ◽  
Elisabetta Schiavello ◽  
Monika Warmuth-Metz ◽  
...  
Keyword(s):  
Molecular Markers ◽  
Retrospective Analysis ◽  
Diffuse Intrinsic Pontine Glioma ◽  
Pontine Glioma ◽  
Mri Features

scholarly journals PDCT-16. A PHASE I STUDY OF SUBEROYLANILIDE HYDROXAMIC ACID (SAHA) WITH TEMSIROLIMUS IN CHILDREN WITH NEWLY DIAGNOSED OR PROGRESSIVE DIFFUSE INTRINSIC PONTINE GLIOMA (DIPG)

2019 ◽  
Vol 21 (Supplement_6) ◽  
pp. vi186-vi187
Author(s):  
Soumen Khatua ◽  
Joya Chandra ◽  
Vidya Gopalakrishnan ◽  
Leena Ketonen ◽  
Michael Johnson ◽  
...  
Keyword(s):  
Radiation Therapy ◽  
Phase I ◽  
Dose Escalation ◽  
Survival Data ◽  
Progressive Disease ◽  
Maximum Tolerated Dose ◽  
Diffuse Intrinsic Pontine Glioma ◽  
Newly Diagnosed ◽  
Pontine Glioma ◽  
Phase Ii Studies

Abstract Diffuse intrinsic pontine glioma (DIPG) in children remains essentially incurable with a median survival of 12 months. Radiation therapy is currently the standard treatment modality. To date chemotherapy or biologic agents have had no meaningful increase in survival. Data from biopsied DIPG showed the PI3K/AKT/mTOR pathway as a major oncogenic player. Thus targeting these pathways with mTOR inhibitors could hold promise. Genomic and epigenetic insights have shown the role of histone deacetylase (HDAC) in these tumors. Both temsirolimus (mTOR inhibitor) and SAHA (HDAC inhibitor) are well described in pediatric clinical trials with an established maximum tolerated dose (MTD) for phase II studies. This is the first ongoing phase I multi-targeted therapeutic study, in pediatric DIPG, using SAHA and temsirolimus in a combinatorial approach, targeting the key oncogenic pathway and molecules (NCT02420613 currently recruiting). Patients with a performance score of 50 or greater, with newly diagnosed (Stratum I), or progressive (stratum II) DIPG will be eligible. Biopsy is not mandatory. A standard 3 + 3 design is being used for this study. 1 dose escalation and 2 dose de-escalations are permitted beyond the starting dose level. In stratum I patients receive radiation therapy concurrently with vorinostat on the days of radiation, followed by adjuvant therapy with vorinostat and temsirolimus for 10 cycles if they do not have progressive disease. In stratum II patients receive vorinostat and temsirolimus for 12 cycles (28 day cycle), if they do not have progressive disease. Correlative studies evaluating histone acetylation, phosphorylated 70S6K and Akt are being performed. Currently 5 patients have been enrolled, and the study is at the dose escalation level with no dose limiting toxicity (DLT) seen so far. Safety, adverse effects, biological correlatives, and clinical outcomes will be reported once study is completed.

scholarly journals DIPG-55. PATTERNS OF CEREBROSPINAL FLUID DIVERSION AND SURVIVAL IN CHILDREN WITH DIFFUSE INTRINSIC PONTINE GLIOMA: A REPORT FROM THE INTERNATIONAL DIPG REGISTRY

2020 ◽  
Vol 22 (Supplement_3) ◽  
pp. iii297-iii298
Author(s):  
Tabitha Cooney ◽  
Mariko DeWire-Schottmiller ◽  
Adam Lane ◽  
Raya Saab ◽  
Pratiti Bandopadhayay ◽  
...  
Keyword(s):  
Overall Survival ◽  
Survival Data ◽  
Clinical Symptoms ◽  
Progression Free Survival ◽  
Standard Of Care ◽  
Diffuse Intrinsic Pontine Glioma ◽  
Pontine Glioma ◽  
Exact Test ◽  
Significant Difference ◽  
Csf Diversion

Abstract There is no standard of care for cerebrospinal (CSF) diversion in children with diffuse intrinsic pontine glioma (DIPG), nor understanding of survival impact. We evaluated CSF diversion characteristics in children with DIPG to determine incidence, indications and potential impact on survival. Data was extracted from subjects registered in the International DIPG registry (IDIPGR). IDIPGR team personnel obtained clinical and radiographic data from the registry database and when appropriate, abstracted additional data from individual medical records. Univariable analyses were performed using the Fisher’s exact test or Wilcoxon rank sum test. Survival was estimated using the Kaplan-Meier method. Evaluable patients (n=457) met criteria for DIPG diagnosis by central radiology review. Ninety-two patients (20%) had permanent CSF diversion. Indications for permanent diversion were hydrocephalus (41%), hydrocephalus and clinical symptoms (35%), and clinical symptoms alone (3%). Those with permanent diversion were significantly younger at diagnosis than those without diversion (median 5.3 years vs 6.9 years, p=0.0002), otherwise no significant differences in gender, race, or treatment were found. The progression-free and overall survival of those with permanent CSF diversion compared to those without permanent diversion was 4.5 and 10.9 months vs 6.9 and 11.2 months, respectively (p=0.001, p= 0.4). There was no significant difference in overall survival in patients with or without permanent CSF diversion among a large cohort of DIPG patients. Patients without permanent diversion had significantly prolonged progression free survival compared to those with permanent diversion. The qualitative risks and benefits of permanent CSF diversion need to be further evaluated.

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