AI Article Synopsis

  • Lysosomal acid lipase (LAL) is crucial for breaking down triglycerides and cholesteryl esters in the lysosome, and a deficiency in this enzyme leads to a severe condition known as Wolman disease (WD), which can be fatal within a year if untreated.
  • This study focused on the clinical and genetic profiles of seven WD patients from five families in Egypt, employing advanced sequencing methods to identify genetic variants responsible for the disease.
  • Researchers discovered three disease-causing variants in the LIPA gene, including a novel variant that is likely pathogenic, which can aid in understanding the relationship between genetics and disease symptoms and improve genetic counseling efforts.

Article Abstract

Lysosomal acid lipase (LAL) is a necessary enzyme for the hydrolysis of both triglycerides (TGs) and cholesteryl esters (CEs) in the lysosome. Deficiency of this enzyme encoded by the lipase A (LIPA) gene leads to LAL deficiency (LAL-D). A severe disease subtype of LAL-D is known as Wolman disease (WD), present with diarrhea, hepatosplenomegaly, and adrenal calcification. Untreated patients do not survive more than a year. The aim of this study was to assess the clinical and molecular characterizations of WD patients in Egypt. A total of seven patients (from five unrelated Egyptian families) were screened by targeted next-generation sequencing (NGS), and the co-segregation of causative variants was analyzed using Sanger sequencing. Furthermore, multiple in silico analyses were performed to assess the pathogenicity of the candidate variants. Overall, we identified three diseases causing variants harbored in the LIPA gene. One of these variants is a novel missense variant (NM_000235.4: c.1122 T > G; p. His374Gln), which was classified as a likely pathogenic variant. All variants were predicted to be disease causing using in silico analyses. Our findings expand the spectrum of variants involved in WD which may help to investigate phenotype-genotype correlation and assist genetic counseling. To the best of our knowledge, this is the first clinico-genetic study carried out on Egyptian patients affected with WD.

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Source
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC10517033PMC
http://dx.doi.org/10.1007/s12031-023-02139-6DOI Listing

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