Ustekinumab for pyoderma gangrenosum-like skin ulcerations in late-onset leukocyte adhesion deficiency.
Leukocyte adhesion deficiency
in situ cytokine profiles
pyoderma gangrenosum–like ulcers
ustekinumab therapy
Journal
The journal of allergy and clinical immunology. Global
ISSN: 2772-8293
Titre abrégé: J Allergy Clin Immunol Glob
Pays: United States
ID NLM: 9918453488706676
Informations de publication
Date de publication:
May 2024
May 2024
Historique:
received:
05
06
2023
revised:
19
12
2023
accepted:
20
12
2023
medline:
1
4
2024
pubmed:
1
4
2024
entrez:
1
4
2024
Statut:
epublish
Résumé
Leukocyte adhesion deficiency type 1 (LAD-1) is a congenital immunodeficiency leading to impaired trafficking of neutrophils to inflammation sites. Solitary or multiple pyoderma gangrenosum (PG)-like skin ulcers (PGLUs) have been reported previously in 13 children (aged 0.5-19 years) with LAD-1. Our aim was to report the case of a 10-year-old boy presenting with PGLUs as the first manifestation of LAD-1 treated with ustekinumab. We obtained PGLUs were triggered by cutaneous ringworm infection ( PGLUs, triggered by ringworm infection, can be a late harbinger of LAD-1. Ustekinumab is a safe and effective therapeutic option for patients with LAD-1 and PGLUs while bridging the time until stem cell transplantation.
Sections du résumé
Background
UNASSIGNED
Leukocyte adhesion deficiency type 1 (LAD-1) is a congenital immunodeficiency leading to impaired trafficking of neutrophils to inflammation sites. Solitary or multiple pyoderma gangrenosum (PG)-like skin ulcers (PGLUs) have been reported previously in 13 children (aged 0.5-19 years) with LAD-1.
Objective
UNASSIGNED
Our aim was to report the case of a 10-year-old boy presenting with PGLUs as the first manifestation of LAD-1 treated with ustekinumab.
Methods
UNASSIGNED
We obtained
Results
UNASSIGNED
PGLUs were triggered by cutaneous ringworm infection (
Conclusions
UNASSIGNED
PGLUs, triggered by ringworm infection, can be a late harbinger of LAD-1. Ustekinumab is a safe and effective therapeutic option for patients with LAD-1 and PGLUs while bridging the time until stem cell transplantation.
Identifiants
pubmed: 38560722
doi: 10.1016/j.jacig.2024.100233
pii: S2772-8293(24)00029-8
pmc: PMC10981098
doi:
Types de publication
Journal Article
Langues
eng
Pagination
100233Informations de copyright
© 2024 The Author(s).