Literature DB >> 32103901

Lysosomal Acid Lipase Deficiency: Therapeutic Options.

Gregory M Pastores1, Derralynn A Hughes2.   

Abstract

Lysosomal acid lipase (LAL) deficiency is a metabolic (storage) disorder, encompassing a severe (Wolman disease) and attenuated (Cholesterol ester storage disease) subtype; both inherited as autosomal recessive traits. Cardinal clinical features include the combination of hepatic dysfunction and dyslipidemia, as a consequence of cholesteryl esters and triglyceride accumulation, predominately in the liver and vascular and reticuloendothelial system. Significant morbidity can arise, due to liver failure and/or atherosclerosis; in part related to the severity of the underlying gene defect and corresponding enzyme deficiency. Diagnosis is based on demonstration of decreased LAL enzyme activity, complemented by analysis of the cognate gene defects. Therapeutic options include dietary manipulation and the use of lipid-lowering drugs. Sebelipase alfa, a recombinant enzyme replacement therapy, has garnered regulatory approval, following demonstration of improvements in disease-relevant markers and clinical benefit in clinical trials, which included increased survival in the most severe cases.
© 2020 Pastores and Hughes.

Entities:  

Keywords:  atherosclerosis; dyslipidemia; enzyme replacement therapy; hepatomegaly; lipid-lowering medications; lysosomal acid lipase deficiency; lysosomal storage disease

Year:  2020        PMID: 32103901      PMCID: PMC7023879          DOI: 10.2147/DDDT.S149264

Source DB:  PubMed          Journal:  Drug Des Devel Ther        ISSN: 1177-8881            Impact factor:   4.162


  46 in total

1.  Infantile-onset Pompe disease: A case series highlighting early clinical features, spectrum of disease severity and treatment response.

Authors:  Penny Owens; Melanie Wong; Kaustuv Bhattacharya; Carolyn Ellaway
Journal:  J Paediatr Child Health       Date:  2018-06-11       Impact factor: 1.954

2.  High Sustained Antibody Titers in Patients with Classic Infantile Pompe Disease Following Immunomodulation at Start of Enzyme Replacement Therapy.

Authors:  Esther Poelman; Marianne Hoogeveen-Westerveld; Marian A Kroos-de Haan; Johanna M P van den Hout; Kees J Bronsema; Nico C van de Merbel; Ans T van der Ploeg; W W M Pim Pijnappel
Journal:  J Pediatr       Date:  2018-02-07       Impact factor: 4.406

3.  Update on lysosomal acid lipase deficiency: Diagnosis, treatment and patient management.

Authors:  Carmen Camarena; Luis J Aldamiz-Echevarria; Begoña Polo; Miguel A Barba Romero; Inmaculada García; Jorge J Cebolla; Emilio Ros
Journal:  Med Clin (Barc)       Date:  2017-03-09       Impact factor: 1.725

Review 4.  Lysosomal acid lipase deficiency--an under-recognized cause of dyslipidaemia and liver dysfunction.

Authors:  Željko Reiner; Ornella Guardamagna; Devaki Nair; Handrean Soran; Kees Hovingh; Stefano Bertolini; Simon Jones; Marijana Ćorić; Sebastiano Calandra; John Hamilton; Terence Eagleton; Emilio Ros
Journal:  Atherosclerosis       Date:  2014-04-15       Impact factor: 5.162

5.  Childhood/adult-onset lysosomal acid lipase deficiency: A serious metabolic and vascular phenotype beyond liver disease-four new pediatric cases.

Authors:  Pierre Poinsot; Sophie Collardeau Frachon; Lioara Restier; André Sérusclat; Mathilde Di Filippo; Sybil Charrière; Philippe Moulin; Alain Lachaux; Noel Peretti
Journal:  J Clin Lipidol       Date:  2016-12-06       Impact factor: 4.766

6.  Targeted disruption of the mouse lysosomal acid lipase gene: long-term survival with massive cholesteryl ester and triglyceride storage.

Authors:  H Du; M Duanmu; D Witte; G A Grabowski
Journal:  Hum Mol Genet       Date:  1998-09       Impact factor: 6.150

7.  Molecular and clinical characterization of a series of patients with childhood-onset lysosomal acid lipase deficiency. Retrospective investigations, follow-up and detection of two novel LIPA pathogenic variants.

Authors:  Livia Pisciotta; Giulia Tozzi; Lorena Travaglini; Roberta Taurisano; Tiziano Lucchi; Giuseppe Indolfi; Francesco Papadia; Maja Di Rocco; Lorenzo D'Antiga; Patricia Crock; Komal Vora; Scott Nightingale; Helen Michelakakis; Anastasia Garoufi; Lilia Lykopoulou; Stefano Bertolini; Sebastiano Calandra
Journal:  Atherosclerosis       Date:  2017-08-26       Impact factor: 5.162

8.  Metabolism of Non-Enzymatically Derived Oxysterols: Clues from sterol metabolic disorders.

Authors:  William J Griffiths; Eylan Yutuc; Jonas Abdel-Khalik; Peter J Crick; Thomas Hearn; Alison Dickson; Brian W Bigger; Teresa Hoi-Yee Wu; Anu Goenka; Arunabha Ghosh; Simon A Jones; Douglas F Covey; Daniel S Ory; Yuqin Wang
Journal:  Free Radic Biol Med       Date:  2019-04-19       Impact factor: 7.376

9.  Long term substrate reduction therapy with ezetimibe alone or associated with statins in three adult patients with lysosomal acid lipase deficiency.

Authors:  Maja Di Rocco; Livia Pisciotta; Annalisa Madeo; Marta Bertamino; Stefano Bertolini
Journal:  Orphanet J Rare Dis       Date:  2018-01-27       Impact factor: 4.123

10.  Case series of sebelipase alfa hypersensitivity reactions and successful sebelipase alfa rapid desensitization.

Authors:  Michelle F Huffaker; Anne Y Liu; Gregory M Enns; Suresh Vijay; Antonio J Amor; N Franklin Adkinson
Journal:  JIMD Rep       Date:  2019-08-01
View more
  6 in total

1.  Intracellular-to-extracellular localization switch of acidic lipase in Enterobacter cloacae through multi-objective medium optimization: aqueous two-phase purification and activity kinetics.

Authors:  Atim Asitok; Maurice Ekpenyong; Nkpa Ogarekpe; Richard Antigha; Iquo Takon; Anitha Rao; Juliet Iheanacho; Sylvester Antai
Journal:  World J Microbiol Biotechnol       Date:  2022-10-14       Impact factor: 4.253

2.  Lessons Learned From the Long-Term Use of Enzyme Replacement Therapy in the Treatment of Lysosomal Acid Lipase Deficiency.

Authors:  Alanna Strong; Can Ficicioglu
Journal:  J Pediatr Gastroenterol Nutr       Date:  2022-05-17       Impact factor: 3.288

3.  Diagnosis, treatment, and follow-up of a case of Wolman disease with hemophagocytic lymphohistiocytosis.

Authors:  Federico Baronio; Francesca Conti; Angela Miniaci; Filomena Carfagnini; Valeria Di Natale; Giulio Di Donato; Matthias Testi; Camilla Totaro; Alessandro De Fanti; Sara Boenzi; Carlo Dionisi-Vici; Susanna Esposito; Andrea Pession
Journal:  Mol Genet Metab Rep       Date:  2021-12-20

4.  Therapeutic efficacy of rscAAVrh74.miniCMV.LIPA gene therapy in a mouse model of lysosomal acid lipase deficiency.

Authors:  Patricia Lam; Anna Ashbrook; Deborah A Zygmunt; Cong Yan; Hong Du; Paul T Martin
Journal:  Mol Ther Methods Clin Dev       Date:  2022-08-04       Impact factor: 5.849

Review 5.  A systematic review of economic evaluations of enzyme replacement therapy in Lysosomal storage diseases.

Authors:  Eleni Ioanna Katsigianni; Panagiotis Petrou
Journal:  Cost Eff Resour Alloc       Date:  2022-09-19

6.  Preclinical Enzyme Replacement Therapy with a Recombinant β-Galactosidase-Lectin Fusion for CNS Delivery and Treatment of GM1-Gangliosidosis.

Authors:  Jason Andrew Weesner; Ida Annunziata; Tianhong Yang; Walter Acosta; Elida Gomero; Huimin Hu; Diantha van de Vlekkert; Jorge Ayala; Xiaohui Qiu; Leigh Ellen Fremuth; David N Radin; Carole L Cramer; Alessandra d'Azzo
Journal:  Cells       Date:  2022-08-19       Impact factor: 7.666

  6 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.