Evidence map›Paper›PMID 36683202›Full record

ReviewPediatric blood & cancer2023

Kaposiform lymphangiomatosis: Diagnosis, pathogenesis, and treatment.

C Griffin McDaniel, Denise M Adams, Kimberley E Steele, Adrienne M Hammill, A Carl Merrow, Janet L Crane, Christopher L Smith, Harry P W Kozakewich, Timothy D Le Cras

Open access · hybridAbstract readReview
In one paragraph

Review in Pediatric blood & cancer, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 26 papers.

0numbers the graph read from it
0cells of the map it votes in
26citing papers in PubMed
10.2field-weighted citation impact, top 1% 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.

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

26 citing papers in PubMed, 32 citations in OpenAlex.

  1. Sirolimus outperforms alpelisib in models ofBlood vessels, thrombosis & hemostasis · 2026
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  13. [Lymphatic malformations in childhood and adolescence].Radiologie (Heidelberg, Germany) · 2025
    Article
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  15. Lyve1-Driven NrasPediatric blood & cancer · 2025
    Article
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  17. Review
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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

9 authors at 6 institutions in 1 country.

C Griffin McDanielUniversity of Cincinnati College of Medicine, Cincinnati, Ohio, USA.ORCID 0000-0002-0573-0956
Denise M AdamsDivision of Oncology, Comprehensive Vascular Anomalies Program, Children's Hospital of Philadelphia, University of Pennsylvania Perelman School of Medicine, Philadelphia, Pennsylvania, USA.
Kimberley E SteeleCollaborative Research Advocacy for Vascular Anomalies Network (CaRAVAN), Bethesda, Maryland, USA.ORCID 0000-0001-5096-6549
Adrienne M HammillDivision of Hematology, Cancer and Blood Diseases Institute, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, USA.ORCID 0000-0003-0820-4924
A Carl MerrowDepartment of Radiology and Medical Imaging, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, USA.
Janet L CraneDivision of Pediatric Endocrinology, Department of Pediatrics, Johns Hopkins University School of Medicine, Baltimore, Maryland, USA.
Christopher L SmithDivision of Cardiology, Children's Hospital of Philadelphia, University of Pennsylvania Perelman School of Medicine, Philadelphia, Pennsylvania, USA.
Harry P W KozakewichDepartment of Pathology, Boston Children's Hospital, Harvard Medical School, Boston, Massachusetts, USA.
Timothy D Le CrasDepartment of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, Ohio, USA.ORCID 0000-0001-8538-2703
Cincinnati Children's Hospital Medical Center · USChildren's Hospital of Philadelphia · USBoston Children's Hospital · USCollaborative Group (United States) · USJohns Hopkins University · USUniversity of Cincinnati Medical Center · US

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 life-threatening rare disease that can cause substantial morbidity, mortality, and social burdens for patients and their families. Diagnosis often occurs long after initial symptoms, and there are few centers in the world with the expertise to diagnose and care for patients with the disease. KLA is a lymphatic anomaly and significant advancements have been made in understanding its pathogenesis and etiology since its first description in 2014. This review provides multidisciplinary, comprehensive, and state-of-the-art information on KLA patient presentation, diagnostic imaging, pathology, organ involvement, genetics, and pathogenesis. Finally, we describe current therapeutic approaches, important areas for research, and challenges faced by patients and their families. Further insights into the pathogenesis of KLA may advance our understanding of other vascular anomalies given that similar signaling pathways may be involved.

Indexed as

Lymphatic AbnormalitiesHumansSignal Transductionkaposiform lymphangiomatosislymphatic anomaliesrare diseases

Identifiers

PMID36683202
PMCPMC10018800
OpenAlexW4317717765

What OpenQuestion holds

Textmetadata
LicenceTDM
Read underepoch 390

Registered trials

None linked

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.