Evidence map›Paper›PMID 41691135›Full record

ReviewJournal of neuro-oncology2026

Belzutifan-induced tumor regression in sporadic hemangioblastoma: a case report and literature review.

Rebekka E Hooks, Niket Yadav, Mark Willy L Mondia, Georgios Mantziaris, Alaa Saleh, Anna Vi Jones, Matthew McCord, Melike Mut, Ashok R Asthagiri, Benjamin W Purow

Abstract readCase ReportsReview
In one paragraph

Review in Journal of neuro-oncology, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
–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

1 citing paper in PubMed.

  1. 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

10 authors.

Rebekka E Hooks *Division of Neuro-Oncology, Department of Neurology, University of Virginia, Charlottesville, Virginia, USA.
Niket Yadav *Division of Neuro-Oncology, Department of Neurology, University of Virginia, Charlottesville, Virginia, USA.
Mark Willy L MondiaDivision of Neuro-Oncology, Department of Neurology, University of Virginia, Charlottesville, Virginia, USA.
Georgios MantziarisDepartment of Neurosurgery, University of Virginia, Charlottesville, Virginia, USA.
Alaa SalehDepartment of Radiology and Medical Imaging, University of Virginia, Charlottesville, Virginia, USA.
Anna Vi JonesDepartment of Pathology, University of Virginia, Charlottesville, Virginia, USA.
Matthew McCordDepartment of Pathology, University of Virginia, Charlottesville, Virginia, USA.
Melike MutDepartment of Neurosurgery, University of Virginia, Charlottesville, Virginia, USA.
Ashok R AsthagiriDepartment of Neurosurgery, University of Virginia, Charlottesville, Virginia, USA.
Benjamin W PurowDivision of Neuro-Oncology, Department of Neurology, University of Virginia, Charlottesville, Virginia, USA. BWP5G@uvahealth.org.ORCID http://orcid.org/0000-0001-7372-6062

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundSporadic hemangioblastomas (sHBs) are less prevalent than those in von Hippel-Lindau (VHL) disease. Surgical resection and radiation therapy remain the standard treatments, and medical treatment options for sHBs are limited. While belzutifan shows a favorable response in VHL-associated HBs, its efficacy in sHBs is uncertain. We present a case of probable belzutifan-induced tumor reduction in a patient with sHB. METHODS AND

resultsWe present a case of a 65-year-old man with progressive right-sided hemifacial paresthesia, retro-orbital pain, and periorbital edema, subsequently diagnosed with progressive right trigeminal nerve-associated sHB. The patient did not meet clinical or genetic criteria for VHL disease. Serial magnetic resonance imaging (MRI) demonstrated significant tumor progression over seven years. Treatment included 12.5 Gy of radiation in 2016, subtotal resection in 2021, and re-radiation with 50.6 Gy in 2022. Repeat debulking resection was deemed high-risk due to tumor location, and prior treatment precluded further re-radiation. Following multidisciplinary review and patient preference, medical therapy was pursued, and belzutifan at 120 mg/day was initiated. MRI demonstrated tumor reduction from 3.4 cm to 3.1 cm by month 2. Due to anemia and fatigue, belzutifan was reduced and continued at 80 mg/day with stable hemoglobin above 10.0 and tolerable fatigue. Serial brain MRIs showed a durable tumor reduction, with a −7.00 mm/year median linear growth rate reduction by month 13.

conclusionTo our knowledge, this is the first report of belzutifan treatment for sHB, resulting in tumor reduction. Belzutifan may offer a possible treatment option for residual, progressive, and/or pre-surgical sHBs.

Indexed as

Cerebellar NeoplasmsHemangioblastomaAgedHumansIndenesMagnetic Resonance ImagingMalebelzutifanIndenesBelzutifanCNSHIF-2αSporadic hemangioblastomaVHL

Identifiers

PMID41691135
PMCPMC12906557

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Registered trials

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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.