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Cited by 143Liver Disease and TransplantationLiver Diseases and ImmunityLiver Disease Diagnosis and Treatment

Vascular involvement of the liver in Turner's syndrome

Roulot, Dominique, Degott, Claude, Chazouillères, Olivier, Oberti, Frédéric, Calès, Paul, Carbonell, Nicolas, Benferhat, Said, Bresson-Hadni, Solange +1 more·2004·Hepatology
DOI 10.1002/hep.20026
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Abstract

Unexplained liver test abnormalities are frequent in patients with Turner's syndrome. This cohort study was performed to clarify the histopathologic features, causes, and long-term outcome of liver involvement in these patients. Thirty patients with persistently abnormal liver test results were followed-up for 8.8 +/- 5.2 years. Liver specimens were available in 27 patients. Marked architectural changes were present in 10 patients, including nodular regenerative hyperplasia in six, multiple focal nodular hyperplasia in two, and cirrhosis in two patients. These changes frequently were associated with obliterative portal venopathy lesions and with aortic malformations. There was mild to moderate portal fibrosis in 15 of the 17 other patients, inflammatory infiltrates in nine patients, and nonalcoholic fatty liver disease in 11 patients. Bile duct alterations resembling small duct sclerosing cholangitis were observed in 21 patients (with or without architectural changes). There was no viral, alcoholic, autoimmune, or drug-induced liver damage. Portal hypertension was observed in four patients with marked architectural changes, including three in whom refractory ascites or recurrent variceal bleeding developed, one of whom underwent transplantation. None of the patients without marked architectural changes experienced progressive or decompensated liver disease. There was no evidence of liver toxicity from estrogen replacement therapy. In conclusion, the main causes of liver involvement in Turner's syndrome are vascular disorders, probably of a congenital origin, and nonalcoholic fatty liver disease. In patients with vascular disorders, severe liver disease requiring liver transplantation may develop. Estrogen therapy does not appear to be pathogenically implicated.

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Authors
Roulot, D., Degott, C., Chazouillères, O., Oberti, F., Calès, P., Carbonell, N., Benferhat, S., Bresson-Hadni, S., & Valla, D.
Affiliations
  • Hôpital Jean-Verdier (FR)
  • Hôpital Beaujon (FR)
  • Hôpital Saint-Antoine (FR)
  • Centre Hospitalier Universitaire d'Angers (FR)
Corresponding author
Roulot, Dominique
ORCID
Venue
Hepatology
Publisher
Ovid Technologies (Wolters Kluwer Health)
Published
2004-01
Volume
39
Issue
1
Pages
239-247
ISSN
0270-9139 (ISSN-L)
DOI
10.1002/hep.20026
OpenAlex
W2020025580
Cited by
143
References
43
Citations by year
2026: 2 · 2025: 3 · 2024: 12 · 2023: 8 · 2022: 12 · 2021: 4 · 2020: 7 · 2019: 8 · 2018: 6 · 2017: 3 · 2016: 2 · 2015: 3 · 2014: 1 · 2013: 12 · 2012: 6
FWCI
7.15
Type
journal article
Language
en
Open access
closed
Licence
doi.wiley.com/10.1002/tdm_license_1.1 (tdm, 4261 d)
Locations
  • Hepatology (publishedVersion)
  • PubMed (publishedVersion)
  • HAL (submittedVersion)
Topics
  • Health Sciences › Medicine › Hepatology › Liver Disease and Transplantation
  • Health Sciences › Medicine › Hepatology › Liver Diseases and Immunity
  • Health Sciences › Medicine › Epidemiology › Liver Disease Diagnosis and Treatment
Keywords
Turner syndrome, liver involvement, liver histopathology, non-alcoholic fatty liver disease, portal hypertension, coarctation of the aorta, nodular regenerative hyperplasia, focal nodular hyperplasia, liver cirrhosis, sclerosing cholangitis, estrogen replacement therapy, liver transplantation, variceal bleeding, ascites
MeSH terms
Adult, Aorta, Bilirubin, Biopsy, Esophageal and Gastric Varices, Female, Follow-Up Studies, Humans, Liver Circulation, Liver Diseases, Middle Aged, Portal Vein, Turner Syndrome, Ultrasonography, Cohort Studies, Gallstones

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