Evidence map›Paper›PMID 41788185›Full record

ArticleJournal of vascular anomalies2025

Trametinib normalizes angiopoietin-2 levels and successfully treats kaposiform lymphangiomatosis.

C Griffin McDaniel, Svatava Merkle, Sophia Lacuna, Stephanie Cooper, Rebecca Courtemanche, Tram Nguyen, Timothy D Le Cras

Abstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in Journal of vascular anomalies, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing 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

2 citing papers in PubMed.

  1. RAS Inhibitor RMC-7977 Blocks Vascular Overgrowth of NRASJournal of cellular and molecular medicine · 2026
    Article
  2. 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

7 authors.

C Griffin McDanielDepartment of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, Ohio, USA.
Svatava MerkleDepartment of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, Ohio, USA.
Sophia LacunaThe Kids Clinic, British Columbia Children's Hospital, University of British Columbia, Vancouver, Canada.
Stephanie CooperDivision of Plastic Surgery, British Columbia Children's Hospital, University of British Columbia, Vancouver, Canada.
Rebecca CourtemancheDivision of Plastic Surgery, British Columbia Children's Hospital, University of British Columbia, Vancouver, Canada.
Tram NguyenThe Kids Clinic, British Columbia Children's Hospital, University of British Columbia, Vancouver, Canada.
Timothy D Le CrasDepartment of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, Ohio, USA.

Funding

Pathogenesis and Treatment of Kaposiform LymphangiomatosisR01HL156866 · NHLBI · CINCINNATI CHILDRENS HOSP MED CTR · PI TIMOTHY DAVID LE CRAS · 2022 to 2026
$3.3M
NHLBI NIH HHS R01 HL156866
6 · The paper itself

Abstract

Kaposiform lymphangiomatosis (KLA) is a complex lymphatic anomaly with high morbidity and mortality. Angiopoietin-2 (Ang-2) levels are elevated in the blood of patients with KLA and can be used to aid in diagnosis and monitor treatment response. We report a male child with hydrocele, a left-sided pleural effusion, splenomegaly, and consumptive coagulopathy who was eventually diagnosed with KLA. Sirolimus did not provide a lasting therapeutic response, so treatment was switched to trametinib. The patient had an excellent response to trametinib with resolution of the pleural effusion, splenomegaly, and coagulopathy; normalization of Ang-2 levels; and significant improvement in clinical symptoms.

Indexed as

Complex lymphatic anomalyMEK inhibitionsirolimus

Identifiers

PMID41788185
PMCPMC12959458

What OpenQuestion holds

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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.