Arterial complications in classical Ehlers-Danlos syndrome: a case series.


Journal

Journal of medical genetics
ISSN: 1468-6244
Titre abrégé: J Med Genet
Pays: England
ID NLM: 2985087R

Informations de publication

Date de publication:
11 2020
Historique:
received: 07 11 2019
revised: 14 02 2020
accepted: 17 02 2020
pubmed: 30 5 2020
medline: 9 7 2021
entrez: 30 5 2020
Statut: ppublish

Résumé

The Ehlers-Danlos syndromes (EDS) are a group of connective tissue disorders with several recognised types. Patients with a type of EDS have connective tissue abnormalities resulting in a varying degree of joint hypermobility, skin and vascular fragility and generalised tissue friability. Classical EDS (cEDS) typically occurs as a result of dominant pathogenic variants in A cohort of 154 patients with a clinical diagnosis of cEDS from the UK was analysed. Seven patients (4.5%) with a diagnosis of cEDS (four pathogenic, one likely pathogenic and two variants of uncertain significance in There is a possible increased risk of arterial complications in patients with cEDS, although not well-defined. Clinicians need to be aware of this possibility when presented with a patient with an arterial complication and features of cEDS. Long-term management in families with cEDS and a vascular complication should be individually tailored to the patient's history and their family's history of vascular events.

Sections du résumé

BACKGROUND
The Ehlers-Danlos syndromes (EDS) are a group of connective tissue disorders with several recognised types. Patients with a type of EDS have connective tissue abnormalities resulting in a varying degree of joint hypermobility, skin and vascular fragility and generalised tissue friability. Classical EDS (cEDS) typically occurs as a result of dominant pathogenic variants in
METHODS
A cohort of 154 patients with a clinical diagnosis of cEDS from the UK was analysed.
RESULTS
Seven patients (4.5%) with a diagnosis of cEDS (four pathogenic, one likely pathogenic and two variants of uncertain significance in
CONCLUSION
There is a possible increased risk of arterial complications in patients with cEDS, although not well-defined. Clinicians need to be aware of this possibility when presented with a patient with an arterial complication and features of cEDS. Long-term management in families with cEDS and a vascular complication should be individually tailored to the patient's history and their family's history of vascular events.

Identifiants

pubmed: 32467296
pii: jmedgenet-2019-106689
doi: 10.1136/jmedgenet-2019-106689
doi:

Substances chimiques

COL5A1 protein, human 0
Collagen Type V 0

Types de publication

Case Reports Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

769-776

Informations de copyright

© Author(s) (or their employer(s)) 2020. No commercial re-use. See rights and permissions. Published by BMJ.

Déclaration de conflit d'intérêts

Competing interests: None declared.

Auteurs

Chloe Angwin (C)

National Ehlers-Danlos Syndrome Service, London North West University Healthcare NHS Trust, Harrow, UK.

Angela F Brady (AF)

National Ehlers-Danlos Syndrome Service, London North West University Healthcare NHS Trust, Harrow, UK.

F Michael Pope (FM)

National Ehlers-Danlos Syndrome Service, London North West University Healthcare NHS Trust, Harrow, UK.

Anthony Vandersteen (A)

IWK Health Centre, Maritime Medical Genetics Service, Halifax, Nova Scotia, Canada.

Duncan Baker (D)

Connective Tissue Disorders Service, Sheffield Diagnostic Genetics Service, Sheffield Children's Hospital, Sheffield, UK.

Harveer Cheema (H)

Connective Tissue Disorders Service, Sheffield Diagnostic Genetics Service, Sheffield Children's Hospital, Sheffield, UK.

Glenda Sobey (G)

National Ehlers-Danlos Syndrome Service, Northern General Hospital, Sheffield, UK.

Diana Johnson (D)

National Ehlers-Danlos Syndrome Service, Northern General Hospital, Sheffield, UK.

Kate von Klemperer (K)

Department of Cardiology, Saint Bartholomew's Hospital, London, UK.

Hanadi Kazkaz (H)

Hypermobility Service, Department of Rheumatology, University College London Hospitals NHS Foundation Trust, London, UK.

Fleur van Dijk (F)

National Ehlers-Danlos Syndrome Service, London North West University Healthcare NHS Trust, Harrow, UK.

Neeti Ghali (N)

National Ehlers-Danlos Syndrome Service, London North West University Healthcare NHS Trust, Harrow, UK neeti.ghali@nhs.net.

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Classifications MeSH