Juvenile idiopathic arthritis in Harlequin ichthyosis, a rare combination or the clinical spectrum of the disease? Report of a child treated with etanercept and review of the literature.
Etanercept
Harlequin ichthyosis
Intra-articular corticosteroids injection
Juvenile idiopathic arthritis
Journal
Pediatric rheumatology online journal
ISSN: 1546-0096
Titre abrégé: Pediatr Rheumatol Online J
Pays: England
ID NLM: 101248897
Informations de publication
Date de publication:
03 Jun 2021
03 Jun 2021
Historique:
received:
27
09
2020
accepted:
20
05
2021
entrez:
4
6
2021
pubmed:
5
6
2021
medline:
24
12
2021
Statut:
epublish
Résumé
Harlequin ichthyosis (HI) is the most severe phenotype of autosomal recessive congenital ichthyosis. Juvenile Idiopathic Arthritis (JIA) represents a heterogenous group of disorders all sharing the clinical manifestation of chronic arthritis. Association of HI and chronic arthritis has been reported in few cases. We report the case of a child with HI who developed a severe form of chronic polyarthritis during the first years of life, treated with repeated multiple joint injections, methotrexate and etanercept with good response and without any adverse events. The reported case and the literature review highlighted the presence of a peculiar severe seronegative polyarthritis with early onset in a series of patients with HI, suggesting that polyarthritis may be a specific manifestation of HI, rather than a rare combination of two separate conditions.
Sections du résumé
BACKGROUND
BACKGROUND
Harlequin ichthyosis (HI) is the most severe phenotype of autosomal recessive congenital ichthyosis. Juvenile Idiopathic Arthritis (JIA) represents a heterogenous group of disorders all sharing the clinical manifestation of chronic arthritis. Association of HI and chronic arthritis has been reported in few cases.
CASE PRESENTATION
METHODS
We report the case of a child with HI who developed a severe form of chronic polyarthritis during the first years of life, treated with repeated multiple joint injections, methotrexate and etanercept with good response and without any adverse events.
CONCLUSION
CONCLUSIONS
The reported case and the literature review highlighted the presence of a peculiar severe seronegative polyarthritis with early onset in a series of patients with HI, suggesting that polyarthritis may be a specific manifestation of HI, rather than a rare combination of two separate conditions.
Identifiants
pubmed: 34082764
doi: 10.1186/s12969-021-00571-9
pii: 10.1186/s12969-021-00571-9
pmc: PMC8173856
doi:
Substances chimiques
Antirheumatic Agents
0
Etanercept
OP401G7OJC
Types de publication
Case Reports
Journal Article
Review
Langues
eng
Sous-ensembles de citation
IM
Pagination
80Commentaires et corrections
Type : ErratumIn
Références
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