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Hereditary Hemorrhagic Telangiectasia-A Case Series Experience from a Liver Transplant Center in Romania

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Specialty Radiology
Date 2022 Dec 23
PMID 36552966
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Abstract

Hereditary hemorrhagic telangiectasia (HHT) has significant morbidity due to multiorgan involvement and an unpredictable disease course. We analyzed the data of 14 patients diagnosed with HHT. The case series comprised 14 patients with a median age at presentation of 48 years old (41-74 years). In twelve patients (85.7%), the diagnosis was confirmed by using the Curacao Criteria. The most common reason for admission was epistaxis, with 9 patients (57%) presenting with nosebleed refractory to prolonged self-tamponade. The biochemical abnormalities identified were elevations in AP and gamma-GT; liver synthetic function was generally normal, even though 21% of patients had clinical or imaging findings for cirrhosis. Nosebleeds were the main reason for admission and significantly impacted quality of life through anemia and frequent hospital admissions. However, the visceral manifestations seemed to be more serious. The hepatic arteriovenous malformations (AVMs) appeared to remain asymptomatic or led to minimal changes for the majority of patients; some cases were associated with liver and biliary tract ischemia, necrosis leading to acute liver failure and even death. Hepatic AVMs can also lead to high-output heart failure due to arterio-venous shunting. The most frequent AVM was hepatic artery to hepatic vein, with secondary hepatic vein dilation and hemodynamic consequences.

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References
1.
Buscarini E, Leandro G, Conte D, Danesino C, Daina E, Manfredi G . Natural history and outcome of hepatic vascular malformations in a large cohort of patients with hereditary hemorrhagic teleangiectasia. Dig Dis Sci. 2011; 56(7):2166-78. PMC: 3112486. DOI: 10.1007/s10620-011-1585-2. View

2.
Aseni P, Vertemati M, Minola E, Bonacina E . Massive haemoptysis after living donor liver transplantation. J Clin Pathol. 2003; 56(11):876-8. PMC: 1770121. DOI: 10.1136/jcp.56.11.876. View

3.
Lerut J, Orlando G, Adam R, Sabba C, Pfitzmann R, Klempnauer J . Liver transplantation for hereditary hemorrhagic telangiectasia: Report of the European liver transplant registry. Ann Surg. 2006; 244(6):854-62. PMC: 1856634. DOI: 10.1097/01.sla.0000247258.35406.a4. View

4.
Shovlin C, Guttmacher A, Buscarini E, Faughnan M, Hyland R, Westermann C . Diagnostic criteria for hereditary hemorrhagic telangiectasia (Rendu-Osler-Weber syndrome). Am J Med Genet. 2000; 91(1):66-7. DOI: 10.1002/(sici)1096-8628(20000306)91:1<66::aid-ajmg12>3.0.co;2-p. View

5.
Silvain C, Thevenot T, Colle I, Vilgrain V, Dupuis-Girod S, Buscarini E . Hereditary hemorrhagic telangiectasia and liver involvement: Vascular liver diseases: position papers from the francophone network for vascular liver diseases, the French Association for the Study of the Liver (AFEF), and ERN-rare liver. Clin Res Hepatol Gastroenterol. 2020; 44(4):426-432. DOI: 10.1016/j.clinre.2020.03.008. View