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Neurologic Complications in Hereditary Hemorrhagic Telangiectasia with Pulmonary Arteriovenous Malformations: A Systematic Review.
Agarwal, Joel; LaBranche, Jennifer; Dhillon, Simran; Allison, W Ted; Jeerakathil, Thomas; Vethanayagam, Dilini.
Afiliación
  • Agarwal J; Department of Medicine, University of Alberta, Edmonton, Alberta, Canada.
  • LaBranche J; Departments of Biological Sciences & Medical Genetics, University of Alberta, Edmonton, Alberta, Canada.
  • Dhillon S; Edmonton HHT Center, Edmonton, Alberta, Canada.
  • Allison WT; Department of Medicine, University of Alberta, Edmonton, Alberta, Canada.
  • Jeerakathil T; Edmonton HHT Center, Edmonton, Alberta, Canada.
  • Vethanayagam D; Departments of Biological Sciences & Medical Genetics, University of Alberta, Edmonton, Alberta, Canada.
Can J Neurol Sci ; 50(4): 561-572, 2023 07.
Article en En | MEDLINE | ID: mdl-35686303
ABSTRACT

BACKGROUND:

Hereditary hemorrhagic telangiectasia (HHT) is an autosomal dominant multi-organ condition occurring with a 1 in 3800 prevalence in Alberta. This genetic disorder leads to vascular malformations in different organs including the lungs and brain, commonly affecting pulmonary vasculature leading to pulmonary arteriovenous malformations (PAVMs). PAVMs lead to right-to-left shunts, which may be associated with neurologic complications. We aimed to evaluate and summarize the reported neurologic manifestations of individuals with HHT with pre-existing PAVMs.

METHODS:

We performed a qualitative systematic review to determine available literature on neurological complications among patients with PAVMs and HHT. Published studies included observational studies, case studies, prospective studies, and cohort studies including search terms HHT, PAVMs, and various neurologic complications using MEDLINE and EMBASE.

RESULTS:

A total of 449 manuscripts were extracted including some duplicates of titles, abstracts, and text which were screened. Following this, 23 publications were identified for inclusion in the analysis. Most were case reports (n = 15). PAVMs were addressed in all these articles in association with various neurological conditions ranging from cerebral abscess, ischemic stroke, hemorrhagic stroke, embolic stroke, and migraines.

CONCLUSION:

Although HHT patients with PAVMs are at risk for a variety of neurological complications compared to those without PAVMs, the quality and volume of evidence characterizing this association is low. Individuals with PAVMs have a high prevalence of neurological manifestations such as cerebral abscess, transient ischemic attack, cerebral embolism, hemorrhage, and stroke. Mitigating stroke risk by implementing proper standardized screening techniques for PAVMs is invaluable in preventing increased mortality.
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Malformaciones Arteriovenosas / Telangiectasia Hemorrágica Hereditaria / Absceso Encefálico / Accidente Cerebrovascular Tipo de estudio: Observational_studies / Qualitative_research / Risk_factors_studies / Systematic_reviews Límite: Humans Idioma: En Revista: Can J Neurol Sci Año: 2023 Tipo del documento: Article País de afiliación: Canadá

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Malformaciones Arteriovenosas / Telangiectasia Hemorrágica Hereditaria / Absceso Encefálico / Accidente Cerebrovascular Tipo de estudio: Observational_studies / Qualitative_research / Risk_factors_studies / Systematic_reviews Límite: Humans Idioma: En Revista: Can J Neurol Sci Año: 2023 Tipo del documento: Article País de afiliación: Canadá