Liver transplantation as a treatment for Wilson's disease with neurological presentation: a systematic literature review.
Copper
Liver transplantation
Neurological symptoms
Systematic review
Wilson’s disease
Journal
Acta neurologica Belgica
ISSN: 2240-2993
Titre abrégé: Acta Neurol Belg
Pays: Italy
ID NLM: 0247035
Informations de publication
Date de publication:
Apr 2022
Apr 2022
Historique:
received:
07
10
2021
accepted:
13
01
2022
pubmed:
27
1
2022
medline:
9
4
2022
entrez:
26
1
2022
Statut:
ppublish
Résumé
Wilson's disease (WD) is a potentially treatable, genetic disorder of copper metabolism, with survival similar to healthy populations if controlled. However, in almost 50% of WD patients, neurological symptoms persist despite treatment, and in up to 10% of patients, neurological deterioration is irreversible. International guidelines on WD treatment do not recommend liver transplantation (LT) as a treatment for neurological symptoms in WD. However, such treatment has been assessed in retrospective analyses, case and series reports. We aimed to systematically assess all available evidence on the effectiveness and safety of LT in WD patients with neurological presentation. This systematic literature review was performed according to Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines. Studies were identified by searching the PubMed database (up to 6 April 2021) and by screening reference lists. Based on the systematic literature review, 48 articles were identified, showing outcomes of LT in 302 WD patients with neurological symptoms. Of these patients, major improvement was found in 215 cases (71.2%), with no difference in neurological status before and after LT in 21 cases (6.9%). There were 29 deaths (9.6%), neurological worsening in 24 cases (7.9%), and 13 cases (4.3%) were lost to follow-up. The results suggest that LT is a promising method of WD management in patients with severe, neurological symptoms, particularly if the patient has not responded to pharmacological de-coppering treatment. Further studies of LT in these patients are warranted.
Identifiants
pubmed: 35080708
doi: 10.1007/s13760-022-01872-w
pii: 10.1007/s13760-022-01872-w
pmc: PMC8986686
doi:
Substances chimiques
Copper
789U1901C5
Types de publication
Journal Article
Systematic Review
Langues
eng
Sous-ensembles de citation
IM
Pagination
505-518Informations de copyright
© 2022. The Author(s).
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