Literature DB >> 18174560

Cholesterol ester storage disease with unusual neurological manifestations in two siblings: a report from South India.

P S Bindu1, A B Taly, R Christopher, P V BharatKumar, S Panda, M Netravathi, S Ravishankar, A Mahadevan, T C Yasha, N Gayathri.   

Abstract

Cholesterol ester storage disease is a rare autosomal recessive storage disorder resulting from lysosomal acid lipase deficiency. Two siblings manifested with hepatosplenomegaly, ptosis, and bilateral external ophthalmoplegia. Evaluation revealed hyperlipidemia and bilateral adrenal calcifications. Leukocyte acid lipase levels were significantly low in both the patients, compared with controls, suggesting a diagnosis of cholesterol ester storage disease. Ptosis and external ophthalmoplegia have hitherto not been reported in cholesterol ester storage disease.

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Year:  2007        PMID: 18174560     DOI: 10.1177/0883073807307104

Source DB:  PubMed          Journal:  J Child Neurol        ISSN: 0883-0738            Impact factor:   1.987


  4 in total

1.  Molecular markers of brain cholesterol homeostasis are unchanged despite a smaller brain mass in a mouse model of cholesteryl ester storage disease.

Authors:  Amal A Aqul; Charina M Ramirez; Adam M Lopez; Dennis K Burns; Joyce J Repa; Stephen D Turley
Journal:  Lipids       Date:  2021-10-07       Impact factor: 1.880

Review 2.  Targeting Wolman Disease and Cholesteryl Ester Storage Disease: Disease Pathogenesis and Therapeutic Development.

Authors:  Francis Aguisanda; Natasha Thorne; Wei Zheng
Journal:  Curr Chem Genom Transl Med       Date:  2017-01-30

3.  Effect of a common missense variant in LIPA gene on fatty liver disease and lipid phenotype: New perspectives from a single-center observational study.

Authors:  Andrea Pasta; Paolo Borro; Anna Laura Cremonini; Elena Formisano; Giulia Tozzi; Stefano Cecchi; Raffaele Fresa; Sara Labanca; Afscin Djahandideh; Samir Giuseppe Sukkar; Antonino Picciotto; Livia Pisciotta
Journal:  Pharmacol Res Perspect       Date:  2021-10

Review 4.  Lysosomal acid lipase deficiency in pediatric patients: a scoping review.

Authors:  Camila da Rosa Witeck; Anne Calbusch Schmitz; Júlia Meller Dias de Oliveira; André Luís Porporatti; Graziela De Luca Canto; Maria Marlene de Souza Pires
Journal:  J Pediatr (Rio J)       Date:  2021-05-06       Impact factor: 2.990

  4 in total

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