Literature DB >> 26413163

Management of lipoprotein X and its complications in a patient with primary sclerosing cholangitis.

Eric J Brandt1, Shane M Regnier2, Edward Ky Leung3, Sharon H Chou4, Beverly W Baron3, Helen S Te5, Michael H Davidson6, Robert M Sargis7.   

Abstract

Lipoprotein X (LpX) is an abnormal lipoprotein found in conditions such as lecithin:cholesterol acyltransferase deficiency and cholestatic states (e.g., primary biliary cirrhosis and primary sclerosing cholangitis). Management of severe hypercholesterolemia due to LpX with drugs and physical removal methods is not well established in the literature. A case is discussed of a 51-year-old woman who presented with multiple electrolyte abnormalities, xanthomas and neuropathy found to be secondary to LpX in the setting of primary sclerosing cholangitis. This case highlights that oral medications, including statins, may be insufficient to normalize lipid levels or improve clinical symptoms of LpX and presents therapeutic plasma exchange as a safe and effective therapeutic option to treat the morbid sequela of LpX hyperlipidemia.

Entities:  

Keywords:  cholestasis; hypercholesterolemia; lipoprotein X; neuropathy; plasma exchange; primary sclerosing cholangitis; statin; xanthoma

Year:  2015        PMID: 26413163      PMCID: PMC4578808          DOI: 10.2217/clp.15.23

Source DB:  PubMed          Journal:  Clin Lipidol        ISSN: 1758-4302


  36 in total

1.  Identification of the abnormal cholestatic lipoprotein (LP-X) in familial lecithin:Cholesterol acyltransferase deficiency.

Authors:  Harald Torsvik; Kåre Berg; Harry N. Magnani; Walter J. McConathy; Peter Alaupovic; Egil Gjone
Journal:  FEBS Lett       Date:  1972-08-01       Impact factor: 4.124

2.  Effect of lipoprotein-X on hepatic cholesterol synthesis.

Authors:  M Liersch; G Baggio; C C Heuck
Journal:  Atherosclerosis       Date:  1977-04       Impact factor: 5.162

3.  The relationship between serum lipids and the electrophoretic pattern, with particular reference to patients with primary biliary cirrhosis.

Authors:  H G KUNKEL; E H AHRENS
Journal:  J Clin Invest       Date:  1949-11       Impact factor: 14.808

4.  Severe hypercholesterolemia mediated by lipoprotein X in patients with chronic graft-versus-host disease of the liver.

Authors:  A Turchin; D A Wiebe; E W Seely; T Graham; W Longo; R Soiffer
Journal:  Bone Marrow Transplant       Date:  2005-01       Impact factor: 5.483

5.  Multiple lipoprotein and electrolyte laboratory artifacts caused by lipoprotein X in obstructive biliary cholestasis secondary to pancreatic cancer.

Authors:  Tharsan Sivakumar; Sushela Chaidarun; Hong Kee Lee; Mark Cervinski; Richard Comi
Journal:  J Clin Lipidol       Date:  2011-05-18       Impact factor: 4.766

6.  Serum lipids in primary sclerosing cholangitis.

Authors:  Emmanouil Sinakos; Ghulam Abbas; Roberta A Jorgensen; Keith D Lindor
Journal:  Dig Liver Dis       Date:  2011-09-03       Impact factor: 4.088

7.  Amelioration of circulating lipoprotein profile and proteinuria in a patient with LCAT deficiency due to a novel mutation (Cys74Tyr) in the lid region of LCAT under a fat-restricted diet and ARB treatment.

Authors:  Shokichi Naito; Mariko Kamata; Masako Furuya; Miyuki Hayashi; Masayuki Kuroda; Hideaki Bujo; Kouju Kamata
Journal:  Atherosclerosis       Date:  2013-03-14       Impact factor: 5.162

8.  Lipoprotein-X reduces LDL atherogenicity in primary biliary cirrhosis by preventing LDL oxidation.

Authors:  Po-Yuan Chang; Shao-Chun Lu; Ta-Chen Su; San-Fang Chou; Wen-Huei Huang; Joel D Morrisett; Chu-Huang Chen; Chiau-Suong Liau; Yuan-Teh Lee
Journal:  J Lipid Res       Date:  2004-08-16       Impact factor: 5.922

9.  Role of plasma and liver cholesterol- and lipoprotein-metabolism determinants in LpX formation in the mouse.

Authors:  Ignacio Bravo; Ludwig Amigo; David E Cohen; Flavio Nervi; Attilio Rigotti; Omar Francone; Silvana Zanlungo
Journal:  Biochim Biophys Acta       Date:  2007-03-01

10.  Recombinant human LCAT normalizes plasma lipoprotein profile in LCAT deficiency.

Authors:  Sara Simonelli; Cristina Tinti; Laura Salvini; Laura Tinti; Alice Ossoli; Cecilia Vitali; Vitor Sousa; Gaetano Orsini; Maria Luisa Nolli; Guido Franceschini; Laura Calabresi
Journal:  Biologicals       Date:  2013-10-18       Impact factor: 1.856

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  4 in total

1.  Detection of Lipoprotein X (LpX): A challenge in patients with severe hypercholesterolaemia.

Authors:  Agnieszka Ćwiklińska; Agnieszka Mickiewicz; Robert Kowalski; Barbara Kortas-Stempak; Agnieszka Kuchta; Krzysztof Mucha; Michał Makowiecki; Anna Gliwińska; Krzysztof Lewandowski; Leszek Pączek; Marcin Fijałkowski; Marcin Gruchała; Maciej Jankowski
Journal:  J Med Biochem       Date:  2020-09-02       Impact factor: 3.402

2.  Case report: Unusual and extremely severe lipoprotein X-mediated hypercholesterolemia in extrahepatic pediatric cholestasis.

Authors:  Rossella Colantuono; Chiara Pavanello; Andrea Pietrobattista; Marta Turri; Paola Francalanci; Marco Spada; Pietro Vajro; Laura Calabresi; Claudia Mandato
Journal:  Front Pediatr       Date:  2022-08-04       Impact factor: 3.569

3.  LCAT protects against Lipoprotein-X formation in a murine model of drug-induced intrahepatic cholestasis.

Authors:  Marcelo J A Amar; Lita A Freeman; Takafumi Nishida; Maureen L Sampson; Milton Pryor; Boris L Vaisman; Edward B Neufeld; Sotirios K Karathanasis; Alan T Remaley
Journal:  Pharmacol Res Perspect       Date:  2019-12-29

4.  Monocyte phenotyping and management of lipoprotein X syndrome.

Authors:  Zeqin Lian; Anum Saeed; Xueying Peng; Xiaoyuan Dai Perrard; Xiaoming Jia; Aliza Hussain; Christie M Ballantyne; Huaizhu Wu
Journal:  J Clin Lipidol       Date:  2020-09-04       Impact factor: 4.766

  4 in total

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