Literature DB >> 10787452

Massive hepatic fibrosis in Gaucher's disease: clinico-pathological and radiological features.

R H Lachmann1, D G Wight, D J Lomas, N C Fisher, J P Schofield, E Elias, T M Cox.   

Abstract

Hepatomegaly is frequent in patients with type 1 Gaucher's disease and is associated with infiltration of the liver with pathological macrophages. Most patients suffer no significant clinical consequences, but a few develop portal hypertension which may progress to parenchymal liver failure. We describe four patients with Gaucher's disease who have developed portal hypertension. We have reviewed their clinical histories and all available histological and radiological material. All had severe Gaucher's disease with multi-organ involvement, and had undergone splenectomy in childhood. Histologically, this advanced liver disease was characterized by a picture of extreme and advanced confluent fibrosis occupying the central region of the liver. This massive fibrosis is associated with characteristic radiological appearances. The liver histology in these cases is highly unusual and virtually unknown in other conditions. Our studies indicate that without specific treatment the liver disease is progressive and rapidly fatal. However, institution of enzyme replacement therapy with imiglucerase may have beneficial effects even when the condition is far advanced.

Entities:  

Mesh:

Substances:

Year:  2000        PMID: 10787452     DOI: 10.1093/qjmed/93.4.237

Source DB:  PubMed          Journal:  QJM        ISSN: 1460-2393


  25 in total

1.  Gaucher disease gene GBA functions in immune regulation.

Authors:  Jun Liu; Stephanie Halene; Mei Yang; Jameel Iqbal; Ruhua Yang; Wajahat Z Mehal; Wei-Lien Chuang; Dhanpat Jain; Tony Yuen; Li Sun; Mone Zaidi; Pramod K Mistry
Journal:  Proc Natl Acad Sci U S A       Date:  2012-06-04       Impact factor: 11.205

2.  Gaucher disease in the liver on hepatocyte specific contrast agent enhanced MR imaging.

Authors:  Rama S Ayyala; Lisa A Teot; Jeanette M Perez Rossello
Journal:  Pediatr Radiol       Date:  2017-02-01

3.  The risk of Parkinson's disease in type 1 Gaucher disease.

Authors:  Gilberto Bultron; Katherine Kacena; Daniel Pearson; Michael Boxer; Ruhua Yang; Swati Sathe; Gregory Pastores; Pramod K Mistry
Journal:  J Inherit Metab Dis       Date:  2010-02-23       Impact factor: 4.982

4.  Hepatobiliary quiz-10 (2014).

Authors:  Swastik Agrawal; Radha K Dhiman
Journal:  J Clin Exp Hepatol       Date:  2014-06

5.  Aberrant progranulin, YKL-40, cathepsin D and cathepsin S in Gaucher disease.

Authors:  Yuliya Afinogenova; Jiapeng Ruan; Ruhua Yang; Nathaniel Kleytman; Gregory Pastores; Andrew Lischuk; Pramod K Mistry
Journal:  Mol Genet Metab       Date:  2019-07-23       Impact factor: 4.797

Review 6.  Gaucher disease.

Authors:  Aabha Nagral
Journal:  J Clin Exp Hepatol       Date:  2014-04-21

7.  Pulmonary vascular disease in Gaucher disease: clinical spectrum, determinants of phenotype and long-term outcomes of therapy.

Authors:  Sarah Michelman Lo; Jun Liu; F Chen; G M Pastores; J Knowles; M Boxer; Kirk Aleck; Pramod K Mistry
Journal:  J Inherit Metab Dis       Date:  2011-03-29       Impact factor: 4.982

Review 8.  Management of portal hypertension in children.

Authors:  Roberto Gugig; Philip Rosenthal
Journal:  World J Gastroenterol       Date:  2012-03-21       Impact factor: 5.742

9.  Life expectancy in Gaucher disease type 1.

Authors:  Neal J Weinreb; Patrick Deegan; Katherine A Kacena; Pramod Mistry; Gregory M Pastores; Priscilla Velentgas; Stephan vom Dahl
Journal:  Am J Hematol       Date:  2008-12       Impact factor: 10.047

Review 10.  Management of non-neuronopathic Gaucher disease with special reference to pregnancy, splenectomy, bisphosphonate therapy, use of biomarkers and bone disease monitoring.

Authors:  T M Cox; J M F G Aerts; N Belmatoug; M D Cappellini; S vom Dahl; J Goldblatt; G A Grabowski; C E M Hollak; P Hwu; M Maas; A M Martins; P K Mistry; G M Pastores; A Tylki-Szymanska; J Yee; N Weinreb
Journal:  J Inherit Metab Dis       Date:  2008-05-23       Impact factor: 4.982

View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.