Evidence map›Paper›PMID 36389350›Full record

ArticleFrontiers in pediatrics2022

Case report: Kaposi hemangioendothelioma of the right upper limb with the Kasabach-Merritt phenomenon: A potentially lethal diagnostic challenge.

Levin Belani, Jamari Sapuan, Shalimar Abdullah, Erica Yee Hing, C-Khai Loh, Hamidah Alias

Open access · goldAbstract readCase Reports
In one paragraph

Article in Frontiers in pediatrics, 2022. 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, top 74% of its field
1 · What the graph read from it

What it found

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2 · The registry

The trial behind it

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3 · Its place in the literature

Who cites it

1 citing paper in PubMed, 0 citations in OpenAlex.

  1. Article
4 · The record

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5 · Who and what money

Authors and funding

6 authors at 1 institution in 1 country.

Levin BelaniDepartment of Orthopaedic and Traumatology, UKM Medical Centre, Faculty of Medicine, Universiti Kebangsaan Malaysia, Kuala Lumpur, Malaysia.
Jamari SapuanDepartment of Orthopaedic and Traumatology, UKM Medical Centre, Faculty of Medicine, Universiti Kebangsaan Malaysia, Kuala Lumpur, Malaysia.
Shalimar AbdullahDepartment of Orthopaedic and Traumatology, UKM Medical Centre, Faculty of Medicine, Universiti Kebangsaan Malaysia, Kuala Lumpur, Malaysia.
Erica Yee HingDepartment of Radiology, UKM Medical Centre, Faculty of Medicine, Universiti Kebangsaan Malaysia, Kuala Lumpur, Malaysia.
C-Khai LohPediatric Hematology and Oncology Unit, Department of Pediatrics, UKM Medical Centre, Faculty of Medicine, Universiti Kebangsaan Malaysia, Kuala Lumpur, Malaysia.
Hamidah AliasPediatric Hematology and Oncology Unit, Department of Pediatrics, UKM Medical Centre, Faculty of Medicine, Universiti Kebangsaan Malaysia, Kuala Lumpur, Malaysia.
University Kebangsaan Malaysia Medical Centre · MY

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Kaposi hemangioendothelioma (KHE) is a rare vascular neoplasm that presents usually within the first year of life. Because of its rarity and complexity, there is often a delay in diagnosis. KHE could be associated with a life-threatening consumptive coagulopathy named the Kasabach-Merritt phenomenon (KMP). Here, we present the case of a 2-month-old girl who presented with progressive redness and swelling of her right upper limb over 6 weeks. Multiple health practitioners misdiagnosed her condition as an insect bite, cellulitis, and necrotizing fasciitis and gave treatment accordingly, which proved futile. A full blood count revealed bicytopenia of anemia and thrombocytopenia, a normal coagulation cascade, low fibrinogen, and raised D-Dimer levels. The imaging was suggestive of a high-flow vascular tumor likely to be a KHE. Subsequently, she was started on single-agent oral sirolimus with a dose increment to achieve satisfactory therapeutic levels and was treated for 1 year. She successfully completed the treatment regimen and had only transient hypertriglyceridemia, which resolved upon the completion of treatment. Currently, she is in remission 3 years after treatment. Keeping her case as an example, we would like to highlight the potentially lethal misdiagnosis of KHE with KMP, the importance of an early diagnosis of this condition, and the successful treatment outcome with single-agent sirolimus.

Indexed as

kaposi hemangioendotheliomaKasabach–Merrittsirolimustreatment outcomeupper limb

Identifiers

PMID36389350
PMCPMC9664933
OpenAlexW4308008343

What Socratic holds

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LicenceCC BY
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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 Socratic graph.