Higher prevalence of splenic artery aneurysms in hereditary hemorrhagic telangiectasia: Vascular implications and risk factors.
Adult
Aged
Aneurysm
/ diagnostic imaging
Case-Control Studies
Female
Follow-Up Studies
France
/ epidemiology
Humans
Male
Middle Aged
Multidetector Computed Tomography
Prevalence
Prognosis
Retrospective Studies
Risk Factors
Splenic Artery
/ pathology
Telangiectasia, Hereditary Hemorrhagic
/ complications
Vascular Diseases
/ diagnostic imaging
Journal
PloS one
ISSN: 1932-6203
Titre abrégé: PLoS One
Pays: United States
ID NLM: 101285081
Informations de publication
Date de publication:
2020
2020
Historique:
received:
19
02
2019
accepted:
02
12
2019
entrez:
24
1
2020
pubmed:
24
1
2020
medline:
9
4
2020
Statut:
epublish
Résumé
Splenic artery aneurysm (SAA) is a rare but potentially fatal condition. Rupture results in 25% mortality up to 75% in pregnant women with 95% fetal mortality. Brief reports suggest an increased risk of developing SAA in patients with HHT. We analyzed enhanced multidetector CT data in 186 HHT patients matched (gender and ± 5 year old) with 186 controls. We screened for SAA and recorded diameter of splenic and hepatic arteries and hepatic, pancreatic and splenic parenchymal involvements. We determined by univariate and multivariate analysis, the relationship with age, sex, genetic status, cardiovascular risk factors (CVRF) and visceral involvement. SAA concerned 24.7% of HHT patients and 5.4% of controls, p<0.001. Factors associated with increased risk of SAA in HHT were female gender (p = 0.04, OR = 2.12, IC 95% = 1.03-4.50), age (p = 0.0003, OR = 1.04, 95% CI = 1.02-1.06) and pancreatic parenchymal involvement (p = 0.04, OR = 2.13, 95% CI = 1.01-4.49), but not type of mutation, hepatic or splenic parenchymal involvements, splenic size or splenic artery diameter or CVRF. We found a 4.57 higher rate of SAA in HHT patients without evidence of splenic high output related disease or increased CVRF. These results suggest the presence of a vascular intrinsic involvement. It should lead to screening all HHT patients for SAA. The vasculopathy hypothesis could require a change in management as screening of all systemic arteries and even the aorta and to further research in the field.
Sections du résumé
BACKGROUND
Splenic artery aneurysm (SAA) is a rare but potentially fatal condition. Rupture results in 25% mortality up to 75% in pregnant women with 95% fetal mortality. Brief reports suggest an increased risk of developing SAA in patients with HHT.
METHODS
We analyzed enhanced multidetector CT data in 186 HHT patients matched (gender and ± 5 year old) with 186 controls. We screened for SAA and recorded diameter of splenic and hepatic arteries and hepatic, pancreatic and splenic parenchymal involvements. We determined by univariate and multivariate analysis, the relationship with age, sex, genetic status, cardiovascular risk factors (CVRF) and visceral involvement.
RESULTS
SAA concerned 24.7% of HHT patients and 5.4% of controls, p<0.001. Factors associated with increased risk of SAA in HHT were female gender (p = 0.04, OR = 2.12, IC 95% = 1.03-4.50), age (p = 0.0003, OR = 1.04, 95% CI = 1.02-1.06) and pancreatic parenchymal involvement (p = 0.04, OR = 2.13, 95% CI = 1.01-4.49), but not type of mutation, hepatic or splenic parenchymal involvements, splenic size or splenic artery diameter or CVRF.
CONCLUSIONS
We found a 4.57 higher rate of SAA in HHT patients without evidence of splenic high output related disease or increased CVRF. These results suggest the presence of a vascular intrinsic involvement. It should lead to screening all HHT patients for SAA. The vasculopathy hypothesis could require a change in management as screening of all systemic arteries and even the aorta and to further research in the field.
Identifiants
pubmed: 31971937
doi: 10.1371/journal.pone.0226681
pii: PONE-D-19-04950
pmc: PMC6977744
doi:
Types de publication
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Pagination
e0226681Déclaration de conflit d'intérêts
The authors have declared that no competing interests exist.
Références
Dig Dis Sci. 2011 Jul;56(7):2166-78
pubmed: 21290179
Am J Med Genet. 2000 Mar 6;91(1):66-7
pubmed: 10751092
Surgery. 1982 Jun;91(6):694-9
pubmed: 7079972
Am J Surg. 1995 Jun;169(6):580-4
pubmed: 7771620
J Hepatol. 2007 Mar;46(3):499-507
pubmed: 17239481
Int J Cardiol. 2014 Oct 20;176(3):1414-6
pubmed: 25150474
Radiology. 2010 Feb;254(2):479-84
pubmed: 20093519
J Vasc Surg. 1986 May;3(5):836-40
pubmed: 3701947
Int J Surg. 2008 Jun;6(3):261-5
pubmed: 17869597
Surgeon. 2010 Aug;8(4):223-31
pubmed: 20569943
Middle East J Anaesthesiol. 2004 Oct;17(6):1135-42
pubmed: 15651520
Am J Obstet Gynecol. 1993 Jun;168(6 Pt 1):1810-1; discussion 1811-3
pubmed: 8317525
Blood Rev. 2010 Nov;24(6):203-19
pubmed: 20870325
Angiology. 1976 Apr;27(4):223-40
pubmed: 1053528
Ann Vasc Surg. 2002 Jul;16(4):442-9
pubmed: 12089631
J Med Genet. 2011 Feb;48(2):73-87
pubmed: 19553198
J Radiol. 2010 Nov;91(11 Pt 1):1103-11
pubmed: 21178873
Ann Vasc Surg. 2000 May;14(3):223-9
pubmed: 10796953
Radiology. 1968 Jun;90(6):1143-9
pubmed: 5656734
Angiogenesis. 2015 Oct;18(4):511-24
pubmed: 26391603
Lancet. 2004 Mar 13;363(9412):852-9
pubmed: 15031030
Am J Med Genet. 1989 Mar;32(3):291-7
pubmed: 2729347
Bull Soc Int Chir. 1970 Jul-Aug;29(4):210-8
pubmed: 5483639
Am J Med Genet A. 2006 Mar 1;140(5):463-70
pubmed: 16470787
J Vasc Res. 2009;46(2):119-37
pubmed: 18765947
Gut. 1960 Dec;1:312-20
pubmed: 13688586