Literature DB >> 21291321

Wolman disease (LIPA p.G87V) genotype frequency in people of Iranian-Jewish ancestry.

Yadira Valles-Ayoub1, Saghi Esfandiarifard, Daniel No, Pedram Sinai, Zeshan Khokher, Melody Kohan, Tanaz Kahen, Daniel Darvish.   

Abstract

Wolman disease (WD) is a rare inherited condition caused by lysosomal acid lipase (LAL) deficiency first described in Iranian-Jewish (IJ) children. Newborns with WD are healthy and active, but soon the infant develops symptoms of severe malnutrition in the first few months of life, and often dies before the age of 1 year. Harmful amounts of lipids accumulate in the spleen, liver, bone marrow, intestine, adrenal glands, and lymph nodes. Although worldwide incidence is estimated at 1/350,000 newborns, WD occurs at higher than expected frequency in the IJ community of the Los Angeles area. As a validation study, we analyzed 162 DNA specimens of IJ origin by automated sequencing. For LIPA p.G87V (ggc>gtc, alternative numbering p.G66V), a heterozygous frequency of 5/162 (3.086%) was discovered. Thus, we estimate that as high as 1 in 4200 newborns of IJ couples may be at risk. Additional studies are required to confirm and further validate the higher frequencies seen in our sample pool, and to determine if people of IJ and even possibly Middle Eastern descent are at a higher risk for WD.

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Year:  2011        PMID: 21291321     DOI: 10.1089/gtmb.2010.0203

Source DB:  PubMed          Journal:  Genet Test Mol Biomarkers        ISSN: 1945-0257


  6 in total

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Journal:  Rev Endocr Metab Disord       Date:  2018-03       Impact factor: 6.514

2.  Functional Characterization of LIPA (Lysosomal Acid Lipase) Variants Associated With Coronary Artery Disease.

Authors:  Trent D Evans; Xiangyu Zhang; Reece E Clark; Arturo Alisio; Eric Song; Hanrui Zhang; Muredach P Reilly; Nathan O Stitziel; Babak Razani
Journal:  Arterioscler Thromb Vasc Biol       Date:  2019-10-24       Impact factor: 8.311

Review 3.  Novel treatment options for lysosomal acid lipase deficiency: critical appraisal of sebelipase alfa.

Authors:  Kim Su; Emma Donaldson; Reena Sharma
Journal:  Appl Clin Genet       Date:  2016-10-17

Review 4.  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

5.  Lysosomal Acid Lipase Deficiency, a Rare Pathology: The First Pediatric Patient Reported in Colombia.

Authors:  Verónica Botero; Victor H Garcia; Catalina Gomez-Duarte; Ana M Aristizabal; Ana M Arrunategui; Gabriel J Echeverri; Harry Pachajoa
Journal:  Am J Case Rep       Date:  2018-06-09

6.  Sebelipase alfa enzyme replacement therapy in Wolman disease: a nationwide cohort with up to ten years of follow-up.

Authors:  Tanguy Demaret; Florence Lacaille; Camille Wicker; Jean-Baptiste Arnoux; Juliette Bouchereau; Claire Belloche; Cyril Gitiaux; David Grevent; Christine Broissand; Dalila Adjaoud; Marie-Thérèse Abi Warde; Dominique Plantaz; Soumeya Bekri; Pascale de Lonlay; Anaïs Brassier
Journal:  Orphanet J Rare Dis       Date:  2021-12-14       Impact factor: 4.123

  6 in total

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