Evidence map›Paper›PMID 39196581›Full record

ReviewClinical cancer research : an official journal of the American Association for Cancer Research2024

Update on Pediatric Cancer Surveillance Recommendations for Patients with Neurofibromatosis Type 1, Noonan Syndrome, CBL Syndrome, Costello Syndrome, and Related RASopathies.

Melissa R Perrino, Anirban Das, Sarah R Scollon, Sarah G Mitchell, Mary-Louise C Greer, Marielle E Yohe, Jordan R Hansford, Jennifer M Kalish, Kris Ann P Schultz, Suzanne P MacFarland and 17 more

Abstract readReview
In one paragraph

Review in Clinical cancer research : an official journal of the American Association for Cancer Research, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 19 papers, 3 of them syntheses that pooled it.

0numbers the graph read from it
0cells of the map it votes in
19citing papers in PubMed, 3 pooled it
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

19 citing papers in PubMed, 3 syntheses or guidelines pooled it.

  1. Pooled it
  2. Pooled it
  3. Cancer in Multilineage Mosaic RASopathies due to Pathogenic Variants in HRAS or KRAS: A Systematic Review and Meta-analysis.Clinical cancer research : an official journal of the American Association for Cancer Research · 2024
    Pooled it
  4. Article
  5. Advances in the management of pediatric low-grade glioma.Neurotherapeutics : the journal of the American Society for Experimental NeuroTherapeutics · 2026
    Review
  6. Article
  7. Article
  8. Article
  9. Review
  10. Article
  11. Review
  12. Article
  13. Review
  14. Pediatric Cancer Predisposition and Surveillance Update: Summary Perspective and Future Directions.Clinical cancer research : an official journal of the American Association for Cancer Research · 2025
    Review
  15. A literature review and pooled case analysis of cardiofaciocutaneous syndrome to estimate cancer risk.Genetics in medicine : official journal of the American College of Medical Genetics · 2025
    Review
  16. Lifelong Management of Neurofibromatosis 1 Patients.Journal of Korean Neurosurgical Society · 2025
    Article
  17. Review
  18. Article
  19. [Progress in diagnosis and treatment ofZhongguo dang dai er ke za zhi = Chinese journal of contemporary pediatrics
    Review
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

27 authors.

Melissa R PerrinoDepartment of Oncology, St Jude Children's Research Hospital, Memphis, Tennessee.ORCID 0000-0002-0598-7102
Anirban DasDivision of Paediatric Haematology and Oncology, Hospital for Sick Children, University of Toronto, Toronto, Ontario, Canada.ORCID 0000-0001-7653-9529
Sarah R ScollonDepartment of Pediatrics, Texas Children's Cancer and Hematology Center, Baylor College of Medicine, Houston, Texas.ORCID 0000-0003-2017-0245
Sarah G MitchellDepartment of Pediatrics, Children's Healthcare of Atlanta, Aflac Cancer and Blood Disorders Center, Emory University School of Medicine, Atlanta, Georgia.ORCID 0000-0003-1656-2799
Mary-Louise C GreerDepartment of Diagnostic and Interventional Radiology, The Hospital for Sick Children, Toronto, Ontario, Canada.ORCID 0000-0001-6159-5472
Marielle E YoheCenter for Cancer Research, National Cancer Institute, Bethesda, Maryland.ORCID 0000-0001-5356-9426
Jordan R HansfordMichael Rice Centre for Hematology and Oncology, Women's and Children's Hospital, Adelaide, South Australia, Australia.ORCID 0000-0001-7733-383X
Jennifer M KalishDivision of Genetics and Center for Childhood Cancer Research Children's Hospital of Philadelphia, Philadelphia, Pennsylvania.ORCID 0000-0003-1500-9713
Kris Ann P SchultzCancer and Blood Disorders, Children's Minnesota, Minneapolis, Minnesota.ORCID 0000-0002-1788-5832
Suzanne P MacFarlandDivision of Oncology, Department of Pediatrics, Perelman School of Medicine, Children's Hospital of Philadelphia, University of Pennsylvania, Philadelphia, Pennsylvania.ORCID 0000-0001-5894-6403
Wendy K KohlmannVA Medical Center, National TeleOncology Clinical Cancer Genetics Service, Durham, North Carolina.ORCID 0000-0002-9134-9640
Philip J LupoDivision of Hematology-Oncology, Department of Pediatrics, Baylor College of Medicine, Houston, Texas.ORCID 0000-0003-0978-5863
Kara N MaxwellDivision of Oncology, Department of Pediatrics, Perelman School of Medicine, Children's Hospital of Philadelphia, University of Pennsylvania, Philadelphia, Pennsylvania.ORCID 0000-0001-8192-4202
Stefan M PfisterDivision Pediatric Neurooncology, Hopp Children´s Cancer Center Heidelberg (KiTZ), German Cancer Research Center (DKFZ) and German Cancer Consortium (DKTK), Heidelberg University Hospital and National Center for Tumor Diseases (NCT), Heidelberg, Germany.ORCID 0000-0002-5447-5322
Rosanna WeksbergDivision of Clinical and Metabolic Genetics, Department of Pediatrics, Hospital for Sick Children and University of Toronto, Toronto, Ontario, Canada.ORCID 0000-0002-6501-4150
Orli MichaeliDivision of Hematology and Oncology, Schneider Children's Medical Center of Israel, Petach Tikva, Israel.ORCID 0000-0002-6097-6457
Marjolijn C J JongmansPrincess Máxima Center for Pediatric Oncology, Utrecht, the Netherlands.ORCID 0000-0001-6572-4374
Gail E TomlinsonDivision of Hematology-Oncology, Department of Pediatrics, Greehey Children's Cancer Research Institute, University of Texas Health Science Center at San Antonio, San Antonio, Texas.ORCID 0000-0003-2579-1833
Jack BrzezinskiDivision of Paediatric Haematology and Oncology, Hospital for Sick Children, University of Toronto, Toronto, Ontario, Canada.ORCID 0000-0002-9475-0077
Uri TaboriDivision of Paediatric Haematology and Oncology, Hospital for Sick Children, University of Toronto, Toronto, Ontario, Canada.ORCID 0000-0002-5019-2683
Gina M NeyDivision of Cancer Epidemiology and Genetics, National Cancer Institute, National Institutes of Health, Rockville, Maryland.ORCID 0000-0002-6530-780X
Karen W GrippDivision of Medical Genetics, Nemours Children's Hospital, Wilmington, Delaware.ORCID 0000-0001-8200-1733
Andrea M GrossPediatric Oncology Branch, Center for Cancer Research, National Cancer Institute, Bethesda, Maryland.ORCID 0000-0003-1646-4531
Brigitte C WidemannPediatric Oncology Branch, Center for Cancer Research, National Cancer Institute, Bethesda, Maryland.ORCID 0000-0002-9198-7175
Douglas R StewartDivision of Cancer Epidemiology and Genetics, National Cancer Institute, National Institutes of Health, Rockville, Maryland.ORCID 0000-0001-8193-1488
Emma R WoodwardUniversity of Manchester and Manchester Centre for Genomic Medicine, Manchester, United Kingdom.ORCID 0000-0002-6297-2855
Christian P KratzHannover Medical School, Hannover, Germany.ORCID 0000-0003-4120-5873

Funding

Genetic Modifiers of Cancer RiskZIACP010142 · NCI · DIVISION OF CANCER EPIDEMIOLOGY AND GENETICS · PI MIRABELLO, LISA · 2009 to 2025
$17.1M
American Lebanese Syrian Associated Charities (ALSAC)Division of Cancer Epidemiology and Genetics (DCEG)Intramural NIH HHS ZIA CP010142Manchester Biomedical Research Centre (BRC) NIHR203308
6 · The paper itself

Abstract

Neurofibromatosis type 1 (NF1), Noonan syndrome, and related syndromes, grouped as RASopathies, result from dysregulation of the RAS-MAPK pathway and demonstrate varied multisystemic clinical phenotypes. Together, RASopathies are among the more prevalent genetic cancer predisposition syndromes and require nuanced clinical management. When compared with the general population, children with RASopathies are at significantly increased risk of benign and malignant neoplasms. In the past decade, clinical trials have shown that targeted therapies can improve outcomes for low-grade and benign neoplastic lesions but have their own challenges, highlighting the multidisciplinary care needed for such individuals, specifically those with NF1. This perspective, which originated from the 2023 American Association for Cancer Research Childhood Cancer Predisposition Workshop, serves to update pediatric oncologists, neurologists, geneticists, counselors, and other health care professionals on revised diagnostic criteria, review previously published surveillance guidelines, and harmonize updated surveillance recommendations for patients with NF1 or RASopathies.

Indexed as

Costello SyndromeNeoplasmsNeurofibromatosis 1Noonan SyndromeChildGenetic Predisposition to DiseaseHumansras Proteinsras Proteins

Identifiers

PMID39196581
PMCPMC11530332

What OpenQuestion holds

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Read underepoch 390

Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.