Evidence map›Paper›PMID 38657450›Full record

ArticleGynecologic oncology2024

Outcomes in ovarian Sertoli-Leydig cell tumor: A report from the International Pleuropulmonary Blastoma/DICER1 and Ovarian and Testicular Stromal Tumor Registries.

Alexander T Nelson, Anne K Harris, Dave Watson, Junne Kamihara, Kenneth S Chen, Jennifer N Stall, Kyle M Devins, Robert H Young, Damon R Olson, Paige H R Mallinger and 15 more

Registry-linked trialOpen access · greenAbstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in Gynecologic oncology, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to trial NCT01970696 (International Ovarian & Testicular Stromal Tumor Registry), which is not on this map. Cited by 12 papers.

0numbers the graph read from it
0cells of the map it votes in
12citing papers in PubMed
8.0field-weighted citation impact, top 2% of its field
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.

NCT01970696 recruitingnot on this map

International Ovarian & Testicular Stromal Tumor Registry

Typeobservational_patient_registrySponsorChildren's Hospitals and Clinics of MinnesotaRan2011 to 2030Enrolled300ConditionsOvarian Sex-cord Stromal Tumor, Testicular Stromal Tumors, Ovarian Small Cell Carcinoma
3 · Its place in the literature

Who cites it

12 citing papers in PubMed, 14 citations in OpenAlex.

  1. Article
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  8. Article
  9. Prognostic Significance of GermlineJCO precision oncology · 2025
    Article
  10. Review
  11. Update on Pediatric Surveillance Recommendations for PTEN Hamartoma Tumor Syndrome, DICER1-Related Tumor Predisposition, and Tuberous Sclerosis Complex.Clinical cancer research : an official journal of the American Association for Cancer Research · 2025
    Review
  12. DICER1-Related Tumor Predisposition: Identification of At-risk Individuals and Recommended Surveillance Strategies.Clinical cancer research : an official journal of the American Association for Cancer Research · 2024
    Article
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

25 authors at 16 institutions in 3 countries.

Alexander T NelsonInternational Pleuropulmonary Blastoma/DICER1 Registry, Children's Minnesota, Minneapolis, MN, USA; International Ovarian and Testicular Stromal Tumor Registry, Children's Minnesota, Minneapolis, MN, USA; Cancer and Blood Disorders, Children's Minnesota, Minneapolis, MN, USA; University of Minnesota Medical School, Minneapolis, MN, USA.
Anne K HarrisInternational Pleuropulmonary Blastoma/DICER1 Registry, Children's Minnesota, Minneapolis, MN, USA; International Ovarian and Testicular Stromal Tumor Registry, Children's Minnesota, Minneapolis, MN, USA; Cancer and Blood Disorders, Children's Minnesota, Minneapolis, MN, USA.
Dave WatsonResearch Institute, Children's Minnesota, Minneapolis, MN, USA.
Junne KamiharaPediatric Oncology, Dana-Farber/Boston Children's Cancer and Blood Disorders Center, Harvard Medical School, Boston, MA, USA.
Kenneth S ChenDepartment of Pediatrics, UT Southwestern Medical Center, Dallas, TX, USA; Children's Medical Center Research Institute, UT Southwestern Medical Center, Dallas, TX, USA.
Jennifer N StallHospital Pathology Associates, Minneapolis, MN, USA.
Kyle M DevinsJames Homer Wright Pathology Laboratories, Massachusetts General Hospital, Harvard Medical School, Boston, MA, USA.
Robert H YoungJames Homer Wright Pathology Laboratories, Massachusetts General Hospital, Harvard Medical School, Boston, MA, USA.
Damon R OlsonDepartment of Pathology and Laboratory Medicine, Children's Minnesota, Minneapolis, MN, USA.
Paige H R MallingerInternational Pleuropulmonary Blastoma/DICER1 Registry, Children's Minnesota, Minneapolis, MN, USA; International Ovarian and Testicular Stromal Tumor Registry, Children's Minnesota, Minneapolis, MN, USA; Cancer and Blood Disorders, Children's Minnesota, Minneapolis, MN, USA.
Sarah G MitchellAflac Cancer and Blood Disorders Center, Children's Healthcare of Atlanta, Emory University School of Medicine, Atlanta, GA, USA.
Lindsey M HoffmanCenter for Cancer and Blood Disorders, Phoenix Children's Hospital, Phoenix, AZ, USA.
Gail HallidayDepartment of Paediatric Oncology, Great North Children's Hospital, Newcastle-upon-Tyne, UK.
Amina M SuleymanovaInstitute of Pediatric Oncology, Federal State Budgetary Institution N.N. Blokhin National Medical Research Center of Oncology of the Ministry of Health of the Russian Federation, Moscow, Russia.
Julia L Glade BenderDepartment of Pediatrics, Memorial Sloan Kettering Cancer Center, New York, NY, USA.
Yoav H MessingerInternational Pleuropulmonary Blastoma/DICER1 Registry, Children's Minnesota, Minneapolis, MN, USA; International Ovarian and Testicular Stromal Tumor Registry, Children's Minnesota, Minneapolis, MN, USA; Cancer and Blood Disorders, Children's Minnesota, Minneapolis, MN, USA.
Cynthia E HerzogDivision of Pediatrics, Department of Pediatric Patient Care, The University of Texas MD Anderson Cancer Care Center, Houston, TX, USA.
Amanda L FieldResourcePath LLC, Sterling, VA, USA.
A Lindsay FrazierPediatric Oncology, Dana-Farber/Boston Children's Cancer and Blood Disorders Center, Harvard Medical School, Boston, MA, USA.
Douglas R StewartClinical Genetics Branch, Division of Cancer Epidemiology and Genetics, National Cancer Institute, Rockville, MD, USA.
Louis P DehnerLauren V. Ackerman Laboratory of Surgical Pathology, Department of Pathology and Immunology, Washington University School of Medicine, St. Louis, MO, USA.
D Ashley HillResourcePath LLC, Sterling, VA, USA; Lauren V. Ackerman Laboratory of Surgical Pathology, Department of Pathology and Immunology, Washington University School of Medicine, St. Louis, MO, USA.
Deborah F BillmireDivision of Pediatric Surgery, Department of Surgery, Indiana University School of Medicine, Indianapolis, IN, USA.
Dominik T SchneiderClinic of Pediatrics, Municipal Hospital Dortmund, University Witten/Herdecke, Germany.
Kris Ann P SchultzInternational Pleuropulmonary Blastoma/DICER1 Registry, Children's Minnesota, Minneapolis, MN, USA; International Ovarian and Testicular Stromal Tumor Registry, Children's Minnesota, Minneapolis, MN, USA; Cancer and Blood Disorders, Children's Minnesota, Minneapolis, MN, USA. Electronic address: KrisAnn.Schultz@childrensmn.org.
Children's Minnesota · USChildren’s Minnesota - St. Paul Hospital · USDana-Farber/Boston Children's Cancer and Blood Disorders Center · USWashington University in St. Louis · USDivision of Cancer Epidemiology and GeneticsEmory University · USGreat North Children's Hospital · GBHarvard University · USIndiana University – Purdue University Indianapolis · USMassachusetts General Hospital · USMemorial Sloan Kettering Cancer Center · USMinistry of Health of the Russian Federation · RUPhoenix Children's Hospital · USSouthwestern Medical Center · USThe University of Texas MD Anderson Cancer Center · USUniversity of Minnesota Medical Center · US

Funding

X-RAY CRYSTALLOGRAPHYP30CA008748 · NCI · SLOAN-KETTERING INSTITUTE FOR CANCER RES · PI SELWYN M VICKERS · 1985 to 2026
$347.4M
DICER1 and the Pleuropulmonary Blastoma Family Cancer SyndromeR01CA143167 · NCI · CHILDREN'S RESEARCH INSTITUTE · PI HILL, DANA ASHLEY · 2010 to 2020
$3.7M
Detecting DICER1: A global partnership to cure pediatric lung cancerR37CA244940 · NCI · CHILDREN'S HOSPITALS AND CLINICS · PI Kris Ann Pinekenstein Schultz · 2020 to 2026
$2.4M
NCI NIH HHS P30 CA008748NCI NIH HHS R01 CA143167NCI NIH HHS R37 CA244940
6 · The paper itself

Abstract

objectiveSertoli-Leydig cell tumors (SLCTs) are rare sex cord-stromal tumors, representing <0.5% of all ovarian tumors. We sought to describe prognostic factors, treatment and outcomes for individuals with ovarian SLCT.

methodsIndividuals with SLCT were enrolled in the International Pleuropulmonary Blastoma/DICER1 Registry and/or the International Ovarian and Testicular Stromal Tumor Registry. Medical records were systematically abstracted, and pathology was centrally reviewed when available.

resultsIn total, 191 participants with ovarian SLCT enrolled, with most (92%, 175/191) presenting with FIGO stage I disease. Germline DICER1 results were available for 156 patients; of these 58% had a pathogenic or likely pathogenic germline variant. Somatic (tumor) DICER1 testing showed RNase IIIb hotspot variants in 97% (88/91) of intermediately and poorly differentiated tumors. Adjuvant chemotherapy was administered in 40% (77/191) of cases, and among these, nearly all patients received platinum-based regimens (95%, 73/77), and 30% (23/77) received regimens that included an alkylating agent. Three-year recurrence-free survival for patients with stage IA tumors was 93.6% (95% CI: 88.2-99.3%) compared to 67.1% (95% CI: 55.2-81.6%) for all stage IC and 60.6% (95% CI: 40.3-91.0%) for stage II-IV (p < .001) tumors. Among patients with FIGO stage I tumors, those with mesenchymal heterologous elements treated with surgery alone were at higher risk for recurrence (HR: 74.18, 95% CI: 17.99-305.85).

conclusionMost individuals with SLCT fare well, though specific risk factors such as mesenchymal heterologous elements are associated with poor prognosis. We also highlight the role of DICER1 surveillance in early detection of SLCT, facilitating stage IA resection.

Indexed as

DEAD-box RNA HelicasesOvarian NeoplasmsPulmonary BlastomaRegistriesRibonuclease IIISertoli-Leydig Cell TumorAdolescentAdultAgedChemotherapy, AdjuvantFemaleHumansLung NeoplasmsMaleMiddle AgedSex Cord-Gonadal Stromal TumorsDEAD-box RNA HelicasesDICER1 protein, humanRibonuclease IIIDICER1DICER1-related tumor predispositionGynandroblastomaSertoli-Leydig cell tumorSex cord-stromal tumor

Identifiers

PMID38657450
PMCPMC11216876
OpenAlexW4395045375

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Registered trials

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.