Trial reportAnnals of oncology : official journal of the European Society for Medical Oncology2011
Pilot trial of sunitinib therapy in patients with von Hippel-Lindau disease.
Trial report in Annals of oncology : official journal of the European Society for Medical Oncology, 2011. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to trial NCT00330564 (A Phase 2 Study of SU011248), which is not on this map. Cited by 62 papers, 2 of them syntheses that pooled 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.
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.
A Phase 2 Study of SU011248 (Sunitinib Malate) in Von Hippel-Lindau Syndrome
Who cites it
62 citing papers in PubMed, 2 syntheses or guidelines pooled it, 126 citations in OpenAlex.
- Familial Kidney Cancer: Implications of New Syndromes and Molecular Insights.European urology · 2019Pooled it
- Benefit, Risk, and Outcomes in Drug Development: A Systematic Review of Sunitinib.Journal of the National Cancer Institute · 2016Pooled it
- Belzutifan for von Hippel-Lindau Disease: Pancreatic Lesion Population of the Phase 2 LITESPARK-004 Study.Clinical cancer research : an official journal of the American Association for Cancer Research · 2024Trial
- Belzutifan for Renal Cell Carcinoma in von Hippel-Lindau Disease.The New England journal of medicine · 2021Trial
- Pazopanib in patients with von Hippel-Lindau disease: a single-arm, single-centre, phase 2 trial.The Lancet. Oncology · 2018Trial
- Trial
- Rescuing the Function of Missense-Mutated Tumor SuppressorbioRxiv : the preprint server for biology · 2026Article
- Review
- Review
- Dual Inhibition of HIF-1α and HIF-2α as a Promising Treatment for VHL-Associated Hemangioblastomas: A Pilot Study Using Patient-Derived Primary Cell Cultures.Biomedicines · 2025Article
- Case report: Von Hippel-Lindau syndrome with multisystem involvement: a therapeutic dilemma.Frontiers in oncology · 2025Article
- Fibroblast growth factor receptor expression in hemangioblastomas: A novel therapeutic target.PloS one · 2025Article
- New Developments in VHL-Associated Neuroendocrine Neoplasms.Current oncology reports · 2025Review
- Neurosurgical Implications of Targeting Hypoxia-Inducible Factor 2α in Hemangioblastomas with Belzutifan.World neurosurgery · 2024Review
- Spinal cord hemangioblastomas in von Hippel-Lindau disease.Neuro-oncology advances · 2024Review
- Genetics, Pathophysiology, and Current Challenges in Von Hippel-Lindau Disease Therapeutics.Diagnostics (Basel, Switzerland) · 2024Review
- The Role of Radiotherapy, Chemotherapy, and Targeted Therapies in Adult Intramedullary Spinal Cord Tumors.Cancers · 2024Review
- Comprehensive treatment of von Hippel-Lindau disease: A case report.Cancer innovation · 2024Article
- Article
- Belzutifan: a novel therapeutic for the management of von Hippel-Lindau disease and beyond.Future oncology (London, England) · 2024Review
2 more citing papers are in PubMed but not listed here.
Corrections and comments
- Commented on by
Authors and funding
10 authors at 3 institutions in 2 countries.
Funding
Abstract
backgroundVon Hippel-Lindau (VHL) disease induces vascular neoplasms in multiple organs. We evaluated the safety and efficacy of sunitinib in VHL patients and examined the expression of candidate receptors in archived tissue.
methodsPatients with VHL were given four cycles of 50 mg sunitinib daily for 28 days, followed by 14 days off. Primary end point was toxicity. Modified RECIST were used for efficacy assessment. We evaluated 20 archival renal cell carcinomas (RCCs) and 20 hemangioblastomas (HBs) for biomarker expression levels using laser-scanning cytometry (LSC).
resultsFifteen patients were treated. Grade 3 toxicity included fatigue in five patients. Dose reductions were needed in 10 patients. Eighteen RCC and 21 HB lesions were evaluable. Six of the RCCs (33%) responded partially, versus none of the HBs (P = 0.014). LSC revealed that mean levels of phosphorylated vascular endothelial growth factor receptor-2 were lower in HB than in RCC endothelium (P = 0.003) and mean phosphorylated fibroblast growth factor receptor substrate-2 (pFRS2) levels were higher in HB (P = 0.003).
conclusionsSunitinib treatment in VHL patients showed acceptable toxicity. Significant response was observed in RCC but not in HB. Greater expression of pFRS2 in HB tissue than in RCC raises the hypothesis that treatment with fibroblast growth factor pathway-blocking agents may benefit patients with HB.
Indexed as
Identifiers
What OpenQuestion holds
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.