Atherotome Dislodgement and Successful Retrieval During Cutting Balloon Pulmonary Angioplasty

DeSena, Holly C.; Ramaciotti, Claudio; Nugent, Alan

doi:10.1161/circinterventions.112.969899

Cited by 2 publications

(1 citation statement)

References 3 publications

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“…The second most commonly reported structural abnormality was an aneurysm (26/150 patients, 17.3%) (Figure 1B), mostly in the basilary (8/150 patients, 5.3%) 24,32,37,39,58,64 , abdominal (8/150 patients, 5.3%) 31,32,39,49,60,65 and carotid (7/150 patients, 4.7%) 10,24,39,48,66,67 arteries. Vessel agenesis and hypoplasia (10 males vs 6 females, 1.7x more) 24,25,28,29,33,38,41,42,58,[68][69][70][71] and esophageal varices were most reported in male patients (6 males vs 3 females, 2x more) 26,30,67,[72][73][74][75][76][77] and patent ductus arteriosus was more highly reported in female patients (7 females vs 2 males, 3.5x more) 37,38,42,45,63,[78]…”

Section: Resultsmentioning

confidence: 99%

Sex differences and risk factors for bleeding in Alagille syndrome

Hankeová

Hul

Laznovsky

et al. 2021

Preprint

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Alagille syndrome (ALGS) is generally thought of as a pediatric cholestatic liver disease, but spontaneous bleeds are a major cause of death. Here, we investigated bleeding in patients and a mouse model for ALGS (Jag1Ndr/Ndr mice) and asked whether phenotypes identified in mice could be detected in patients non-invasively. Jag1Ndr/Ndr mice spontaneously bled, exhibiting a thin skull and vascular defects. Vascular abnormalities included artery-vein crossings, tortuous vessels, and capillary breakdown. Furthermore, Jag1Ndr/Ndr arteries had sparse vascular smooth muscle cell coverage, which was further aggravated by hypertension. Retinographs from patients with ALGS, biliary atresia, or CADASIL confirmed tortuous blood vessels and artery-vein crossings in ALGS. We also identified sex-specific differences: fewer major vessels in female Jag1Ndr/Ndr mice, and four-fold more female than male patients with intracranial hemorrhage. In conclusion, dysfunctional Jag1 disrupted vascular growth and homeostasis, with sex-specific differences. The mouse model for Alagille syndrome demonstrated multiple bleeding risk factors and vascular defects that can be identified in patients non-invasively.

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