Evidence map›Paper›PMID 39081307›Full record

ReviewFrontiers in immunology2024

Sweet syndrome associated with moderate leukocyte adhesion deficiency type I: a case report and review of the literature.

Yoshine Saito, Anupama Kewalramani, Xiao P Peng, Aimee Magnarelli, Howard M Lederman

Abstract readCase ReportsReview
In one paragraph

Review in Frontiers in immunology, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

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

0 citing papers in PubMed.

No citing paper in PubMed yet.

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

5 authors.

Yoshine SaitoUniversity of Maryland School of Medicine, Baltimore, MD, United States.
Anupama KewalramaniDepartment of Pediatrics, Division of Pulmonology/Allergy, University of Maryland School of Medicine, Baltimore, MD, United States.
Xiao P PengDepartment of Genetic Medicine, Johns Hopkins University School of Medicine, Baltimore, MD, United States.
Aimee MagnarelliEudowood Division of Pediatric Allergy, Immunology and Rheumatology, Johns Hopkins University School of Medicine, Baltimore, MD, United States.
Howard M LedermanEudowood Division of Pediatric Allergy, Immunology and Rheumatology, Johns Hopkins University School of Medicine, Baltimore, MD, United States.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Sweet syndrome is an acute febrile neutrophilic dermatosis characterized by the infiltration of neutrophils into the skin. It may occur idiopathically or be linked to malignancies, inflammatory or autoimmune diseases. Leukocyte adhesion deficiency type I (LAD-I) is an inborn error immunity wherein leukocytes lack adhesion molecules necessary for migration to infection sites due to mutations in the CD18 gene encoding β2 integrins. We present a case of a 16-month-old female initially diagnosed and treated for Sweet syndrome based on histopathological findings with recurrent flare episodes. Subsequent workup revealed LAD-I, making this case the first documented association between Sweet syndrome and LAD-I. Moreover, we reviewed the pertinent literatures detailing the concurrence of neutrophilic dermatosis and immunodeficiency disorders. This case underscores the significance of comprehensive evaluation for Sweet syndrome patients who are refractory to conventional treatments.

Indexed as

Leukocyte-Adhesion Deficiency SyndromeSweet SyndromeCD18 AntigensFemaleHumansInfantMutationNeutrophilsSkinCD18 Antigensinborn error of immunity (IEI)leukocyte adhesion deficiency type 1neutrophilic dermatosissweet syndromewhole exome sequencing

Identifiers

PMID39081307
PMCPMC11286406

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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.