Skin microvascular dysfunction as an early cardiovascular marker in primary hyperoxaluria type I.
Adolescent
Adult
Cardiovascular Diseases
/ diagnosis
Case-Control Studies
Child
Child, Preschool
Conservative Treatment
Endothelium, Vascular
/ physiopathology
Female
Humans
Hyperoxaluria, Primary
/ complications
Kidney Transplantation
Laser-Doppler Flowmetry
Liver Transplantation
Male
Microvessels
/ diagnostic imaging
Muscle, Smooth, Vascular
/ physiopathology
Prospective Studies
Rare Diseases
/ complications
Skin
/ blood supply
Time Factors
Treatment Outcome
Vascular Stiffness
/ physiology
Vasodilation
/ physiology
Young Adult
Cardiovascular risk
Endothelium
Hyperoxaluria type I
Microcirculation
Renal transplantation
Journal
Pediatric nephrology (Berlin, Germany)
ISSN: 1432-198X
Titre abrégé: Pediatr Nephrol
Pays: Germany
ID NLM: 8708728
Informations de publication
Date de publication:
02 2019
02 2019
Historique:
received:
17
04
2018
accepted:
03
09
2018
revised:
03
08
2018
pubmed:
3
10
2018
medline:
3
4
2020
entrez:
3
10
2018
Statut:
ppublish
Résumé
Primary hyperoxaluria type 1 (PH1) is an orphan inborn error of oxalate metabolism leading to hyperoxaluria, progressive renal failure, oxalate deposition, and increased cardiovascular complications. As endothelial dysfunction and arterial stiffness are early markers of cardiovascular risk, we investigated early endothelial and vascular dysfunction in young PH1 patients either under conservative treatment (PH1-Cons) or after combined kidney liver transplantation (PH1-T) in comparison to healthy controls (Cont-H) and patients with a past of renal transplantation (Cont-T). Skin microvascular function was non-invasively assessed by laser Doppler flowmetry before and after stimulation by current, thermal, or pharmacological (nitroprussiate (SNP) or acetylcholine (Ach)) stimuli in young PH1 patients and controls. Seven PH1-Cons (6 F, median age 18.2) and 6 PH1-T (2 F, median age 13.3) were compared to 96 Cont-H (51 F, median age 14.2) and 6 Cont-T (4 F, median age 14.5). The endothelium-independent vasodilatation (SNP) was severely decreased in PH1-T compared to Cont-H. Ach, current-induced vasodilatation (CIV), and thermal response was increased in PH1-Cons and Cont-T compared to controls. PH1-T patients displayed severely decreased smooth muscle capacity to vasodilate. An exacerbated endothelial-dependent vasodilation suggests a role for silent inflammation in the early dysfunction of microcirculation observed in PH1-Cons and Cont-T.
Sections du résumé
BACKGROUND
Primary hyperoxaluria type 1 (PH1) is an orphan inborn error of oxalate metabolism leading to hyperoxaluria, progressive renal failure, oxalate deposition, and increased cardiovascular complications. As endothelial dysfunction and arterial stiffness are early markers of cardiovascular risk, we investigated early endothelial and vascular dysfunction in young PH1 patients either under conservative treatment (PH1-Cons) or after combined kidney liver transplantation (PH1-T) in comparison to healthy controls (Cont-H) and patients with a past of renal transplantation (Cont-T).
METHODS
Skin microvascular function was non-invasively assessed by laser Doppler flowmetry before and after stimulation by current, thermal, or pharmacological (nitroprussiate (SNP) or acetylcholine (Ach)) stimuli in young PH1 patients and controls.
RESULTS
Seven PH1-Cons (6 F, median age 18.2) and 6 PH1-T (2 F, median age 13.3) were compared to 96 Cont-H (51 F, median age 14.2) and 6 Cont-T (4 F, median age 14.5). The endothelium-independent vasodilatation (SNP) was severely decreased in PH1-T compared to Cont-H. Ach, current-induced vasodilatation (CIV), and thermal response was increased in PH1-Cons and Cont-T compared to controls.
CONCLUSIONS
PH1-T patients displayed severely decreased smooth muscle capacity to vasodilate. An exacerbated endothelial-dependent vasodilation suggests a role for silent inflammation in the early dysfunction of microcirculation observed in PH1-Cons and Cont-T.
Identifiants
pubmed: 30276532
doi: 10.1007/s00467-018-4081-5
pii: 10.1007/s00467-018-4081-5
doi:
Types de publication
Controlled Clinical Trial
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Pagination
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