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Clinicopathologic Features, Management, and Outcomes Of Pediatric CRTC1::TRIM11 and MED15::ATF1 Tumors With Spitzoid Morphology: A Case Series

  1. Author:
    Sargen, Michael R [ORCID]
    Fitzhugh, Madeline H [ORCID]
    Ko, Jennifer S
    Close, Allison G [ORCID]
    Horton, Sara C
    Malik, Faizan
    Furtado, Larissa V
    Bahrami, Armita
    Seynnaeve, Brittani K N [ORCID]
    Pappo, Alberto S [ORCID]
    Liu, Vincent
    Cheung, Kevin [ORCID]
    Pack, Svetlana D
    Xi, Liqiang
    Raffeld, Mark
    Lee, Ina
    Gagan, Jeffrey
    Aldape, Kenneth
    Barr, Frederic G
    Hewitt, Stephen M
    Ferrone, Christina K
    Hayden, Anna
    Hatton, Jessica N
    Goldstein, Alisa M
    Pfeiffer, Ruth M
    Widemann, Brigitte
    Malone, Mary Frances Wedekind [ORCID]
    Gross, Andrea M [ORCID]
    Kaplan, Rosandra
    Yohe,Marielle
    Berrebi, Kristen G

  2. Author Address

    Division of Cancer Epidemiology and Genetics, National Cancer Institute, National Institutes of Health, Rockville, Maryland, USA., Department of Dermatology, University of Iowa Hospitals and Clinics, Iowa City, Iowa, USA., Department of Anatomic Pathology, Cleveland Clinic, Cleveland, Ohio, USA., Department of Hematology-Oncology, Helen DeVos Children 39;s Hospital, Grand Rapids, Michigan, USA., Department of Hematology-Oncology, University of Iowa Stead Family Children 39;s Hospital, Iowa City, Iowa, USA., Department of Pathology, St. Jude Children 39;s Research Hospital, Memphis, Tennessee, USA., Department of Pathology and Laboratory Medicine, Emory University School of Medicine, Atlanta, Georgia, USA., Department of Pediatrics, University of Pittsburgh School of Medicine, Pittsburgh, Pennsylvania, USA., Department of Oncology, St. Jude 39;s Children 39;s Research Hospital, Memphis, Tennessee, USA., Laboratory of Pathology, Center for Cancer Research, National Cancer Institute, National Institutes of Health, Bethesda, Maryland, USA., Division of Dermatology, University of Louisville School of Medicine, Louisville, Kentucky, USA., Pediatric Oncology Branch, National Cancer Institute, National Institutes of Health, Bethesda, Maryland, USA., Laboratory of Cell and Developmental Signaling, National Cancer Institute, National Institutes of Health, Frederick, Maryland, USA.,
    1. Year: 2025
    2. Date: May 19
    3. Epub Date: 2025 05 19
  2. Journal: Pediatric Blood & Cancer
    1. Pages: e31814
  4. Type of Article: Article
  5. Article Number: e31814
  1. Abstract:

    Limited information is available on the clinicopathologic features and management of pediatric CRTC1::TRIM11 (CTRTs) and MED15::ATF1-rearranged tumors (MARTs). In this series of pediatric CTRTs (n = 5) and MARTs (n = 2), all tumors exhibited spitzoid morphology and expressed melanocytic markers. Metastatic progression occurred in one CTRT and two MARTs: all three received immunotherapy, resulting in disease control in two patients and one death due to progressive disease. A fourth individual had lymph node micrometastasis that remained stable without progression. This case series highlights the potentially aggressive behavior of these tumors and suggests that immunotherapy may be an effective treatment option for advanced disease. © 2025 The Author(s). Pediatric Blood & Cancer published by Wiley Periodicals LLC. This article has been contributed to by U.S. Government employees and their work is in the public domain in the USA.

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External Sources

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  2. DOI: 10.1002/pbc.31814
  3. PMID: 40390195

Library Notes

  1. Fiscal Year: FY2024-2025
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