Targeted DNA Sequencing for Tailored Therapies in Children with Extracranial Solid Tumors

Next-generation sequencing (NGS) is instrumental for clinical decisions on molecularly targeted therapy (TT). In pediatric oncology, TT is a relatively rare choice administered chiefly on a tumor-agnostic basis. The investigation enrolled 304 pediatric patients with extracranial solid tumors that were diagnosed and treated in 2018-2023. Tumor DNA was sequenced using a customized QiaSeq panel (Qiagen, Hilden, Germany) of genes known to be relevant for pediatric solid tumors, including <i>ALK</i>, <i>BRAF</i>, <i>BRCA1/2</i>, <i>EGFR</i>, <i>FGFR1</i>, <i>KIT</i>, <i>MAP2K1/2</i>, <i>NF1</i>, <i>PDGFRA/B</i>, <i>PIK3CA</i>, <i>PTEN</i>, <i>PTPN11</i>, RAS family genes, etc. The assay allowed detection of nucleotide substitutions and small insertions/deletions, as well as gene copy number alterations. TT sensitivity predictors were identified in 120/304 cases (39.5%): Tier II in 83 patients, Tier IB in 32 patients (almost always ALK in neuroblastoma, <i>n</i> = 31) and Tier IA in 5 patients: BRAF p.V600E (<i>n</i> = 3) and NF1 aberrations (<i>n</i> = 2). TT commenced in 21/304 cases (6.9%), often first-line or as a first relapse therapy (14/21 cases), combined with chemotherapy (TT-CT) in 13 cases. The median of TT duration was 10.9 (range 0.8-43.5) months for single-mode and 12.3 (0.3-61.5) months for TT-CT. Clinical benefit rate was achieved in 14/21 patients (66.7%). At the time of writing, nine patients (42.8%) have no progression and are still on treatment for 30.4 months (range 10.3-40.5) after the start of TT. The median time to the best response to TT was 6 (range 0.8-12.3) months. The tolerance was generally good: the therapy was discontinued for toxicity in only one case. The study provides a TT-focused prospective analysis still rare in pediatric oncology. The outcomes indicate satisfactory tolerance and promising efficacy of TT, prompting an update of current treatment standards for several pediatric cancers.

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Journal
International Journal of Molecular Sciences
Published
2025-11-26
DOI
https://doi.org/10.3390/ijms262311463
Primary Topic
Neuroblastoma Research and Treatments
Type
article
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article

Targeted DNA Sequencing for Tailored Therapies in Children with Extracranial Solid Tumors

Natalia Usman, Н. А. Андреева, Н. В. Жуков, D. Yu. Kachanov et al.
International Journal of Molecular Sciences
Neuroblastoma Research and Treatments
article

Targeted DNA Sequencing for Tailored Therapies in Children with Extracranial Solid Tumors

Natalia Usman, Н. А. Андреева, Н. В. Жуков, D. Yu. Kachanov, D. V. Litvinov, G. А. Novichkova, Д. М. Коновалов, Ruslan Abasov, А. Е. Druy, Т. V. Shamanskaya, Nina V. Gegeliya, A. V. Protsvetkina, Nikolay Grachev, N. V. Myakova
article en

Abstract

Next-generation sequencing (NGS) is instrumental for clinical decisions on molecularly targeted therapy (TT). In pediatric oncology, TT is a relatively rare choice administered chiefly on a tumor-agnostic basis. The investigation enrolled 304 pediatric patients with extracranial solid tumors that were diagnosed and treated in 2018-2023. Tumor DNA was sequenced using a customized QiaSeq panel (Qiagen, Hilden, Germany) of genes known to be relevant for pediatric solid tumors, including <i>ALK</i>, <i>BRAF</i>, <i>BRCA1/2</i>, <i>EGFR</i>, <i>FGFR1</i>, <i>KIT</i>, <i>MAP2K1/2</i>, <i>NF1</i>, <i>PDGFRA/B</i>, <i>PIK3CA</i>, <i>PTEN</i>, <i>PTPN11</i>, RAS family genes, etc. The assay allowed detection of nucleotide substitutions and small insertions/deletions, as well as gene copy number alterations. TT sensitivity predictors were identified in 120/304 cases (39.5%): Tier II in 83 patients, Tier IB in 32 patients (almost always ALK in neuroblastoma, <i>n</i> = 31) and Tier IA in 5 patients: BRAF p.V600E (<i>n</i> = 3) and NF1 aberrations (<i>n</i> = 2). TT commenced in 21/304 cases (6.9%), often first-line or as a first relapse therapy (14/21 cases), combined with chemotherapy (TT-CT) in 13 cases. The median of TT duration was 10.9 (range 0.8-43.5) months for single-mode and 12.3 (0.3-61.5) months for TT-CT. Clinical benefit rate was achieved in 14/21 patients (66.7%). At the time of writing, nine patients (42.8%) have no progression and are still on treatment for 30.4 months (range 10.3-40.5) after the start of TT. The median time to the best response to TT was 6 (range 0.8-12.3) months. The tolerance was generally good: the therapy was discontinued for toxicity in only one case. The study provides a TT-focused prospective analysis still rare in pediatric oncology. The outcomes indicate satisfactory tolerance and promising efficacy of TT, prompting an update of current treatment standards for several pediatric cancers.

International Journal of Molecular SciencesVol. 26(23)
Institute of Medical Cell Technologies (RU), Dmitry Rogachev National Research Center of Pediatric Hematology, Oncology and Immunology (RU)
Openalex Percentile: Top 49%
Neuroblastoma Research and Treatments
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